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PubMed2000

Indeterminate fibrohistiocytic lesions of the skin: is there a spectrum between dermatofibroma and dermatofibrosarcoma protuberans?

Routine histology and immunohistochemistry can usually distinguish dermatofibroma (DF) and dermatofibrosarcoma protuberans (DFSP). DF generally expresses factor XIIIa whereas DFSP generally expresses CD34. The authors report 10 cutaneous fibrohistiocytic lesions combining clinical, histologic, and immunohistochemical features of both DF and DFSP. The lesions had an average size of 1.2 cm (range, 0.4-2.7 cm), and occurred on the trunk (n = 6), extremities (n = 3), and face (n = 1) of four men and six women (average age, 30.6 yrs; age range, 15-50 yrs). Eight lesions exhibited acanthosis and densely cellular fascicles with focal storiform areas. All had keloidal collagen, infiltrated the subcutis in a honeycomb pattern, and had low mitotic counts (0 to 4 mitoses per square millimeter). All were diffusely immunoreactive for factor XIIIa (30%-60% of the neoplastic cells) as well as CD34 (20%-70%). This series raises the possibility of a biologic spectrum between DF and DFSP; however, double-immunolabeling studies showed no notable coexpression of factor XIIIa and CD34 by individual cells, suggesting coexistence of two different cellular populations. After an average follow up of 22.3 months (range, 10-46 mos) in six cases, a single recurrence was documented. The ambiguous histologic features and the potential for local recurrence suggest that performing a complete excision may be prudent in these diagnostically indeterminate lesions.

Acanthosis Nigricans↗
PubMed1994

Noncraniofacial manifestations of Crouzon's disease.

Although most interest centers on the craniofacial region in Crouzon's disease, noncraniofacial manifestations are important because they may complicate both diagnosis and management. A population of 59 patients with Crouzon's disease was reviewed to determine the frequency of these deformities. Stylohyoid ligament calcification (50%) and cervical spine (40%) and elbow (18%) abnormalities were the most common. Minor hand deformities (10%), other musculoskeletal deformities (7%), and visceral anomalies (7%) were also seen. Acanthosis nigricans was not present in this population. Recommendations are made for the assessment and management of Crouzon's disease with reference to these areas.

Acanthosis Nigricans↗
PubMed2000

Nonalcoholic steatohepatitis in children.

BACKGROUND: Nonalcoholic steatohepatitis occurs commonly in adults with obesity or diabetes mellitus. There are only a few reports of this condition in children. METHODS: Prospective consecutive clinical series. RESULTS: Between December 1985 and April 1995, 36 children (21 boys, 15 girls) were diagnosed with nonalcoholic steatohepatitis at the Hospital for Sick Children, Toronto. The median age at diagnosis was 12 years (range, 4-16 years). Most patients were referred because of elevated serum aminotransferases or abnormal hepatic sonogram. Thirty patients (83%) were obese. Two patients had diabetes mellitus at diagnosis, and it developed later in two. Fifteen patients had palpable hepatomegaly, and one of these had splenomegaly. None had physical signs of chronic liver disease. Thirteen of 36 patients had acanthosis nigricans. Serum aminotransferases were elevated in all but one patient. Tests for Wilson disease and chronic hepatitis B and C were negative. Serum lipid profiles were abnormal in 18 patients: 7 had hypercholesterolemia, and 11 had hypertriglyceridemia. Twenty-four of 31 examined had abnormal liver sonograms suggestive of fatty infiltration. Twenty-four patients underwent percutaneous liver biopsy: all showed large-droplet fat. Inflammation was present in 88% and fibrosis-cirrhosis in 75%. One 10-year-old patient had established cirrhosis at diagnosis. CONCLUSIONS: Nonalcoholic steatohepatitis occurs in children, is clinically diverse, and may not be benign.

Acanthosis Nigricans↗
PubMed1999

Insulin receptor antibodies and insulin resistance.

The presence of insulin receptor antibodies is a rare cause of insulin resistance. Patients usually have a combination of hyperglycemia, insulin resistance, acanthosis nigricans, and autoimmune features. We report a patient with systemic lupus erythematosus and severe insulin resistance due to insulin receptor antibodies. The most striking aspect of the clinical presentation is the resistance to insulin therapy, with our patient unresponsive to doses of up to 154,075 units in a day. While on a low-dose glucocorticoid therapy, the patient had clinical improvement, and glucose levels subsequently became normal even without insulin and glucocorticoid.

Acanthosis Nigricans↗
PubMed2004

Clinical profiles, occurrence, and management of adolescent patients with HAIR-AN syndrome.

The syndrome of hyperandrogenism, insulin resistance, and acanthosis nigricans (HAIR-AN) is a subphenotype of the polycystic ovary syndrome. It is one of the most common causes of menstrual problems, hyperandrogenic symptoms, and insulin resistance among young women. Review of clinical data in an outpatient adolescent clinic showed that of the 1,002 young women (ages 10-21 years) attending the clinic over a 2-year period, 50 (5%) were diagnosed with HAIR-AN syndrome. Mean age of the patients was 15.5, initial mean weight at diagnosis was 94.5 kg, and the mean BMI was 33.33 kg/m2. Patients were treated with a weight-stabilization and -reduction program, oral contraceptive pills, and in most cases metformin. Of the patients, 80% were compliant with the follow-up and treatment regimen, 60% maintained or reduced their weight, 95% had regular menstrual cycles, and in most patients, the acne and/or hirsutism were the same or better than at the start of treatment. We conclude that HAIR-AN syndrome is a common disease in young women and multifaceted, aggressive treatment appears to be effective in reducing the severity of symptoms and preventing further consequences.

Abnormalities, Multiple↗
PubMed1997

Topical application of viral vectors for epidermal gene transfer.

Efficient gene transfer with extended gene expression is essential for successful treatment of skin diseases using gene therapy. Previously we evaluated a physical gene transfer method (gene gun delivery) for its ability to transfect the epidermis in vivo. In this study, we tested two viral vectors for their ability to transduce murine epidermis through topical application. Both an adenoviral vector and a herpes simplex virus (HSV) amplicon vector transduced murine epidermis with high efficiency after topical application. Differences in amount and duration of transgene expression were compared between these two vectors. Quantitative analysis of reporter lacZ gene expression showed that the viral vector-mediated gene transfers were superior to gene-gun delivery of plasmid DNA. Significant necrosis and cytotoxicity, however, were observed in the HSV-treated skin. In addition, we show that murine epidermis developed hyperkeratosis and acanthosis 4 d after an adenoviral vector containing a human TGF-alpha expression unit was applied topically. Finally we demonstrate the feasibility of transduction of fetal skin in utero by intraamniotic injection of an adenovirus vector.

Acanthosis Nigricans↗
PubMed1989

Heterogeneity of the polycystic ovary syndrome: clinical, endocrine and ultrasound features in 556 patients.

This paper reports an analysis of the clinical, endocrine and ultrasound data within a population of 556 patients with ultrasound-diagnosed polycystic ovaries. Compared with those not so affected, hirsutism was associated with a higher mean serum testosterone concentration, infertility was associated with higher mean gonadotrophin concentrations, obesity was associated with a higher mean serum testosterone concentration, hyperprolactinaemia was associated with a lower mean serum testosterone concentration and smaller ovaries, alopecia was associated with lower mean serum LH and testosterone concentrations, and acanthosis nigricans was associated with obesity and a raised mean serum testosterone concentration. The heterogeneity illustrates the limitations in the use of specific clinical or endocrine criteria as requirements for the diagnosis of the polycystic ovary syndrome.

Acanthosis Nigricans↗
PubMed1997

Terra firma-forme dermatosis.

Terra firma-forme dermatosis (TFFD) is a relatively common but usually unrecognized clinical problem. The cause is unknown. The changes usually occur on the neck in childhood as dirty brown patches that cannot be removed with soap but are easily removed with alcohol. Terra firma-forme dermatosis may be mistaken for acanthosis nigricans.

Acanthosis Nigricans↗
PubMed1998

Partial face-sparing lipodystrophy (Köbberling-Dunnigan syndrome): report of a sporadic case.

A sporadic case is presented of a 20-year-old female with partial face-sparing lipodystrophy, also known as the Köbberling-Dunnigan syndrome with the Type 1 clinical phenotype. This disorder affects the limbs and spares the face. The trunk is spared in the Type 1 phenotype but is affected in the Type 2 phenotype. The patient had the associated features of this syndrome including the hyperinsulinaemia, acanthosis nigricans and mildly raised triglycerides, but not diabetes mellitus.

Acanthosis Nigricans↗
PubMed1993

Insulin requirements in lipodystrophic diabetes.

A ten-fold increase in daily insulin requirements during the administration of total parenteral nutrition (TPN) is described in a patient with congenital generalized lipodystrophy, insulin-requiring diabetes mellitus, and acanthosis nigricans during an episode of acute pancreatitis secondary to hypertriglyceridaemia. After a period of 13 days on TPN, insulin requirements increased dramatically to an average of 1428 units per day for a period of 12 days, to achieve a mean blood glucose level of 10.9 mmol l-1. When the patient resumed feeding and the TPN was discontinued, the average daily insulin requirement was 104 units with a mean 24 h blood glucose of 11.8 mmol l-1. Parenteral administration of energy substrates in a rare case of diabetes mellitus and congenital lipodystrophy complicated by acute pancreatitis resulted in a severe insulin insensitive state due to the combination of the hypermetabolism conferred by the pancreatitis plus transient impairments of the glucose disposal mechanism by the energy substrates provided.

Acanthosis Nigricans↗
PubMed2006

Porokeratosis of Mibelli induced by topical corticosteroid.

BACKGROUND: [corrected] Prorokeratosis of Mibelli is a chronic disorder characterised by slightly atrophic plaques surrounded by keratotic border. METHODS AND RESULTS: A 45-year-old with clobetazole propionate ointment for psoriasis over 15 years developed characteristic lesions of porokeratosis Mibelli on the elbows. Histopathological examination revealed the cornoid lamellae located on the edges of the specimen and psoriasiform acanthosis and a spongiotic pustule formation in the center of the specimen; thus, it was suggested as 'psoriasis encircled by porokeratosis'. CONCLUSIONS: Development of porokeratosis may be explained by the local immunosuppressive effect of the prolonged application of the topical steroid.

Acanthosis Nigricans↗
PubMed2006

Is confluent and reticulated papillomatosis without papillomatosis early or late stage of confluent and reticulated papillomatosis?

The histologic findings of confluent and reticulated papillomatosis include papillomatosis, hyperkeratosis, and minimal or no acanthosis. We present a patient with the clinical findings of confluent and reticulated papillomatosis who responded dramatically to azithromycin, but histologic examination did not reveal papillomatosis. We observed invagination of basket-weave hyperkeratosis through the epidermis. We considered this finding may give a clue for the diagnosis of confluent and reticulated papillomatosis.

Acanthosis Nigricans↗
PubMed1975

Congenital generalized lipodystrophy.

Six patients with congenital generalized lipodystrophy are described. They had generalized paucity of fat tissue, acanthosis nigricans, prominent superficial veins and muscle hypertrophy. They were mentally retarded. Three had corneal opacities. They had normal external genitalia and none was tall for age. Their bone age was advanced and some had minor skeletal anomalies and nephromegaly. The muscle histology on light microscopy was normal. The majority had elevated serum aldolase and to a lesser degree serum lactic dehydrogenase and creatinine phosphokinase. Four of five examined had a myopathic electromyogram. They had normal or deranged liver function tests. The fatty liver infiltration in one seems to be progressive. Four had a normal and two an abnormal metyrapone test. They had an age-dependent abnormality of growth hormone, insulin and carbohydrate homeostasis.

Acanthosis Nigricans↗
PubMed1999

Premature adrenarche: findings in prepubertal African-American and Caribbean-Hispanic girls.

Premature adrenarche was previously thought to be a benign condition. However, the authors and several other research groups have noted hyperinsulinism and insulin resistance in many girls with premature adrenarche. African-American and Caribbean-Hispanic girls with premature adrenarche are frequently obese with marked hyperandrogenism, signs which correlate with the degree of insulin resistance (i.e., those girls who are obese and insulin resistant tend to have higher levels of adrenocorticotropic hormone-stimulated androgens). Also, girls with premature adrenarche and reduced insulin sensitivity can have subtle decreases in their high-density lipoprotein (HDL) profile. Many of these girls have a strong family history of type 2 diabetes mellitus. Preliminary data regarding long-term follow-up of girls with premature adrenarche indicate that those girls who remain obese are at risk of developing polycystic ovary syndrome (PCOS). The term 'syndrome X' refers to the constellation of laboratory and clinical findings associated with hyperinsulinism stemming from insulin resistance. These findings include obesity, acanthosis nigricans, glucose intolerance, type 2 diabetes mellitus, dyslipidaemia with reduced HDL and elevated low-density lipoprotein, cardiovascular disease and PCOS. Hence, for certain girls, premature adrenarche may be a part of the clinical spectrum of syndrome X.

Acanthosis Nigricans↗
PubMed1991

Unusual forms of insulin resistance.

Insulin resistance is an important feature in the pathogenesis of non-insulin-dependent diabetes mellitus. Some rare patients have syndromes associated with a severe form of insulin resistance, usually involving acanthosis nigricans and, in premenopausal females, hyperandrogenism. This review discusses two of the causes of insulin resistance: mutations in the insulin receptor gene and autoantibodies directed against the insulin receptor.

Acanthosis Nigricans↗
PubMedDate not supplied

Type a syndrome of insulin resistance: anterior chamber anomalies of the eye and effects of insulin-like growth factor-I on the retina.

BACKGROUND: The purpose of this work was to describe the anterior chamber and iris anomalies as well as to evaluate the effects of recombinant human insulin-like growth factor-I (rhIGF-I) on the retinal vessels in 2 diabetic patients with type A syndrome of insulin resistance, a rare condition associated with acanthosis nigricans. METHODS: Ophthalmologic examinations, including photographs and fluorescein angiograms, were performed before, and 2 and 4 weeks after starting subcutaneous rhIGF-I treatment, and 3 months after withdrawal of rhIGF-I treatment. RESULTS: Both patients had goniodysgenesis with mild elevation of the intraocular pressure. Before and after 2 weeks of treatment with rhIGF-I, the fundus and the fluorescein angiograms were mainly normal. After 4 weeks of rhIGF-I treatment both patients' retinas revealed leakage of fluorescein. Three (case 1) and 4 months (case 2) after withdrawal of rhIGF-I, the fundus of all four eyes were again without leakage. CONCLUSIONS: The anterior chamber anomalies found in these patients may be part of the type A syndrome of insulin resistance and could alert clinicians that these patients might not have the usual type of diabetes. Moreover, the data show that exogenous rhIGF-I administration in patients with type A syndrome of insulin resistance alters the permeability of the superficial layer of retinal capillaries which is comparable to the earliest angiographic changes in childhood diabetic retinopathy. Whether this is a direct effect of rhIGF-I, as suggested by experiments in an animal model, or an indirect effect due to the near-normalization of the glucose levels by rhIGF-I warrants further investigations. Finally, this work points to an important caveat regarding the therapeutic use of rhIGF-I in this patient population.

Acanthosis Nigricans↗
PubMed2003

Metabolic effects of growth hormone therapy in an Alström syndrome patient.

AIM: To investigate the metabolic effects of recombinant human growth hormone (rhGH) in an Alström syndrome patient with growth hormone deficiency. METHODS: A 15-year-old Alström syndrome boy with growth hormone deficiency received rhGH therapy for 1 year. Biochemical parameters, including hepatic enzyme levels, lipid profiles, and insulin sensitivity, were measured. Body composition analysis and computed tomography scans of the liver were performed. RESULTS: After 1 year of rhGH treatment, body fat mass, fat infiltration in the liver, and serum lipid profiles had all decreased. Insulin sensitivity and acanthosis nigricans improved. CONCLUSION: rhGH therapy might have beneficial effects on body composition, liver fat content, lipid profiles, and insulin resistance in Alström syndrome patients, with improvement of the glucose homeostasis.

Abnormalities, Multiple↗