Costello Syndrome with Severe Nodulocystic Acne: Unexpected Significant Improvement of Acanthosis Nigricans after Oral Isotretinoin Treatment

Sriboonnark, Leelawadee; Arora, Harleen; Falto‐Aizpurua, Leyre; Choudhary, Sonal; Connelly, Elizabeth Alvarez

doi:10.1155/2015/934865

Cited by 5 publications

(5 citation statements)

References 7 publications

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“…Another report was of improvement with sustained maintenance of AN lesions on combination therapy of isotretinoin and metformin 66 . A patient of Costello syndrome showed resolution of AN lesions on neck and hand following treatment by isotretinoin for nodulocystic acne 28 . A case of paraneoplastic AN with gastric adenocarcinoma also showed improvement of lesions after treatment with retinoids 67…”

Section: Systemic Treatmentsmentioning

confidence: 99%

“…The association of AN with rare syndromes has been described: Barter syndrome, 24 Benign encephalopathy, 25 Bloom syndrome, 26 Chondrodystrophy with dwarfism, 27 Costello syndrome, 28 Dermatomyositis, 29 Familial pineal body hypertrophy, 30 Gigantism, 7 Hashimoto thyroiditis, 31 Hirschowitz syndrome, 32 Lipoatrophic diabetes mellitus, 33 Lupoid hepatitis, 34 Lupus erythematosus, 35 Phenylketonuria, 36 Pituitary hypogonadism, 37 Pseudoacromegaly, 38 Prader‐Willi syndrome, 39 Rud syndrome, 40 Scleroderma, 41 Type A syndrome (HAIR‐AN syndrome), 42 Werner syndrome, 43 Wilson syndrome 44 …”

Section: Introductionmentioning

confidence: 99%

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Acanthosis nigricans: A review

Das

Datta²,

Kassir

et al. 2020

J of Cosmetic Dermatology

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Background Acanthosis nigricans (AN) is a cutaneous disorder characterized by symmetric velvety hyperpigmented plaques on intertriginous areas like axilla, neck, inframammary, and groin. Aims To summarize the pathophysiology and classification of AN, provide an update of diagnostic testing strategies, and describe the current therapeutic options described so far in the literature for this disease. Methods A comprehensive english language literature search across multiple databases (PubMed, EMBASE, MEDLINE, and Cochrane) for keywords (alone and in combination) was performed. MeSH as well as non‐MeSH terms such as “acanthosis nigricans,” “classification,” “pathophysiology,” “diagnosis,” “treatment,” “topical drugs,” “systemic drugs,” “chemical peeling,” and “lasers” were taken into consideration. Results The pathophysiology of AN revolves around a multifactorial stimulation of proliferation of epidermal keratinocytes and dermal fibroblasts. Various types of AN include benign, obesity‐associated, syndromic, malignant, acral, unilateral, medication‐induced, and mixed‐type. Homeostasis model assessment‐insulin resistance (HOMA‐IR) is a good tool for assessment of insulin resistance. Management involves general measures (weight reduction and addressing the underlying cause, if any), topical drugs (retinoids, vitamin D analogs, and keratolytics), oral drugs (retinoids and insulin sensitizers), chemical peels (trichloroacetic acid), and lasers (Long pulsed alexandrite, fractional 1550‐nm erbium fiber, and CO2). Conclusion Acanthosis nigricans is a treatable condition; however, complete cure and disappearance of lesions are difficult to achieve. Weight reduction is the most scientific and practical management strategy. Long‐term studies and further research is warranted in the pathophysiology and treatment of this common condition.

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Section: Systemic Treatmentsmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

Acanthosis nigricans: A review

Das

Datta²,

Kassir

et al. 2020

J of Cosmetic Dermatology

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“…Palmoplantar keratoderma is managed with topical tazarotene, urea cream and physical paring. One case report described improvement of acanthosis nigricans after treatment with isotretinoin for nodulocystic acne (Sriboonnark, Aurora, Falto‐Aizpurua, Choudhary, & Connelly, ).…”

Section: Dermatologic Findingsmentioning

confidence: 99%

Costello syndrome: Clinical phenotype, genotype, and management guidelines

Gripp

Morse

Axelrad

et al. 2019

American J of Med Genetics Pt A

111

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Costello syndrome (CS) is a RASopathy caused by activating germline mutations in HRAS. Due to ubiquitous HRAS gene expression, CS affects multiple organ systems and individuals are predisposed to cancer. Individuals with CS may have distinctive craniofacial features, cardiac anomalies, growth and developmental delays, as well as dermatological, orthopedic, ocular, and neurological issues;however, considerable overlap with other RASopathies exists. Medical evaluation requires an understanding of the multifaceted phenotype. Subspecialists may have limited experience in caring for these individuals because of the rarity of CS. Furthermore, the phenotypic presentation may vary with the underlying genotype. These guidelines were developed by an interdisciplinary team of experts in order to encourage timely health care practices and provide medical management guidelines for the primary and specialty care provider, as well as for the families and affected individuals across their lifespan. These guidelines are

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“…Established modalities of treatment of acanthosis nigricans have been topical use of retinoids, α-hydroxy acids, or salicylic acid in adjunct with treatment of underlying condition. [1] Among the orally used drugs, insulin sensitizers such as metformin, rosiglitazone,[16] sitagliptin,[17] and oral retinoids such as isotretinoin and acitretin[18] have been assessed to have some effect in reduction of cutaneous lesions in acanthosis nigricans. However, to the best of our knowledge, effectiveness of alpha-lipoic acid in acanthosis nigricans has not yet been studied.…”

Section: Discussionmentioning

confidence: 99%

Effectiveness and safety of metformin versus Canthex™ in patients with acanthosis nigricans: A randomized, double-blind controlled trial

et al. 2019

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Background: Acanthosis nigricans has been associated with conditions of insulin resistance such as obesity, polycystic ovary syndrome, and type 2 diabetes. Metformin and alpha-lipoic acid, two types of insulin-sensitizing agents, have been demonstrated to reduce insulin levels and improve insulin sensitivity. Alpha-lipoic acid is available as a fixed-dose combination with biotin, calcium pantothenate, and zinc sulfate as Canthex™. Aims: This study aimed to compare the effectiveness, safety, and improvement of the insulin resistance profile of Canthex™ and metformin in acanthosis nigricans. Materials and Methods: In this double-blind, randomized (1:1), active-controlled trial (CTRI/2017/02/007880), participants received either metformin 500 mg BD or Canthex™ BD for 12 weeks. Effectiveness parameters were improvement of severity of neck lesions and neck texture. Serum fasting insulin level, glucose, lipids, body weight, waist circumference, body mass index (BMI), and homeostatic model assessment-insulin resistance (HOMA-IR) were also assessed at baseline and at the end of the study. Adverse effects and changes in routine laboratory parameters were taken as safety parameters. Results: Thirty-three patients were analyzed by modified-intention-to-treat criteria. Severity of neck lesions and texture were comparable at baseline and it showed significant reduction ( P <0.001) in both the treatment arms from the first follow-up onward. No intergroup variation was observed in any of the follow-ups. There was reduction in the values of fasting insulin, blood sugar, total cholesterol, and thyroid-stimulating hormone in both the groups. Weight, BMI, and waist circumference and BMI reduced significantly in both the groups. HOMA-IR decreased significantly in metformin group ( P <0.001). Conclusion: Canthex™ is as effective and safe as metformin in the management of acanthosis nigricans and associated features of insulin resistance.

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Costello Syndrome with Severe Nodulocystic Acne: Unexpected Significant Improvement of Acanthosis Nigricans after Oral Isotretinoin Treatment

Cited by 5 publications

References 7 publications

Acanthosis nigricans: A review

Acanthosis nigricans: A review

Costello syndrome: Clinical phenotype, genotype, and management guidelines

Effectiveness and safety of metformin versus Canthex™ in patients with acanthosis nigricans: A randomized, double-blind controlled trial

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