Letter: Hyfrecation and Curettage as a Treatment for Fibrofolliculomas in Birt?Hogg?Dube Syndrome

Farrant, Paul; Emerson, Russell

doi:10.1111/j.1524-4725.2007.33270.x

Cited by 12 publications

(6 citation statements)

References 4 publications

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“…Surgical and CO2 laser treatments can be used to remove the skin lesions, but the treatment is only temporary and the lesions often return over time. [ 53 , 57 , 59 – 61 ] A recent double-blind placebo-controlled randomized split-face study showed no effect of the topical mTOR inhibitor rapamycin on fibrofolliculoma in BHDS patients. [ 62 ]…”

Section: Resultsmentioning

confidence: 99%

Birt–Hogg–Dubé syndrome: a case report and a review of the literature

Jensen

Villumsen

Skytte

et al. 2017

European Clinical Respiratory Journal

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Background: Birt-Hogg-Dubé syndrome (BHDS) is a rare autosomal dominant inherited syndrome caused by mutations in the folliculin coding gene (FLCN). The clinical manifestations of the syndrome involve the skin, lungs, and kidneys. Because of the rarity of the syndrome, guidelines for diagnosis and management of the patients with BHDS are lacking. Objective: To present a case story and a review of the literature on BHDS in order to give an update on genetics, clinical manifestations, diagnosis, treatment, prognosis and follow-up strategies. Design: Literature review and case story. Results: A PubMed and Embase search identified 330 papers. BHDS is characterized by small benign tumors in the skin, spontaneous pneumothoraces caused by cysts in the lungs and a seven-fold increased risk of renal cancer. A case story of a young female patient presenting with pneumothorax and a family history of recurrent pneumothoraces in many relatives illustrates how the history and the diagnostic work up resulted in a diagnosis of BHDS. Conclusion: BHDS is a rare inherited disorder. In patients with spontaneous pneumothorax or cystic lung disease without any obvious explanation, BHDS should be considered. Concomitant skin manifestations, a family history of familiar pneumothorax, renal cancers and skin manifestations supports the suspicion of BHDS. Early diagnosis is important in order to subject patients to systematic screening for renal cancers. A radiological surveillance strategy for renal cancer is proposed.

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Section: Resultsmentioning

confidence: 99%

Birt–Hogg–Dubé syndrome: a case report and a review of the literature

Jensen

Villumsen

Skytte

et al. 2017

European Clinical Respiratory Journal

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“…However, there has also been evidence of recurrence of facial papules associated with BHD roughly 6 months after treatment with Er:YAG laser reported in another patient [4]. Curettage plus cautery has had promising results for one patient, with only minor recurrence three years after treatment [5]. Shave plus cautery has also been reported to have removed facial lesions successfully in another single case, but there is no mention of patient follow-up [6].…”

Section: Discussionmentioning

confidence: 99%

“…It is simple, safe, cost-effective, and in our case, has shown no evidence of recurrence. In addition, there were no complications of scarring, pigment change, and/or prolonged healing that has been suggested to be associated with other treatment methods, notably laser [5]. Larger numbers of patients need to be treated prior to making any conclusions regarding this treatment method.…”

Section: Discussionmentioning

confidence: 99%

Successful treatment of facial papules with electrodessication in a patient with birt-hogg-dubé syndrome

Pritchard¹,

Mahmoudizad

Parekh³

2014

Dermatology Online Journal

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We report a case of a 51-year-old Hispanic woman who presented with a several year history of multiple flesh colored papules of cosmetic concern on the nose and medial cheeks (Figure 1). Biopsies revealed fibrofolliculoma and trichodiscoma. The patient was referred for genetic testing and was found to be positive for the FLCN gene defect, confirming a diagnosis of Birt-Hogg-Dubé syndrome. Further work-up with screening renal ultrasound and CT scan of the thorax and abdomen was unrevealing. For treatment of these skin lesions, dermasanding was attempted initially with only minimal benefit. She subsequently had multiple lesions treated with hyfrecation (also known as electrodessication or cautery) at a low setting and was very pleased with the results (Figure 2). Importantly, there has yet to be a recurrence of lesions two years status-post hyfrecation.

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“…Various modalities are in use, including excision, laser ablation and curettage. From our own experience and recent literature [41], we suggest that the optimal approach is hyfrecation followed, if necessary, by curettage. This treatment is simple, cheap and can be performed using standard equipment that will be available in most doctors' offices.…”

Section: Treatment Of Bhd Skin Lesionsmentioning

confidence: 96%

What’s new in Birt–Hogg–Dubé syndrome?

Claessens

Vernooij

Luijten

et al. 2012

Expert Review of Dermatology

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Keywords: Birt-Hogg-Dubé • cilia • folliculin • hair follicle tumor • mTOR • planar cell polarity • rapamycin • sebaceous gland • wingless For reprint orders, please contact reprints@expert-reviews.com Expert Review of Dermatology Downloaded from informahealthcare.com by Emory University on 08/10/15 For personal use only. 8 Hasumi Y, Baba M, Ajima R et al. Homozygous loss of BHD causes early embryonic lethality and kidney tumor development with activation of mTORC1 and mTORC2.

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Letter: Hyfrecation and Curettage as a Treatment for Fibrofolliculomas in Birt?Hogg?Dube Syndrome

Cited by 12 publications

References 4 publications

Birt–Hogg–Dubé syndrome: a case report and a review of the literature

Birt–Hogg–Dubé syndrome: a case report and a review of the literature

Successful treatment of facial papules with electrodessication in a patient with birt-hogg-dubé syndrome

What’s new in Birt–Hogg–Dubé syndrome?

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