Bilateral Temporal Triangular Alopecia Associated with Phakomatosis Pigmentovascularis Type IV Successfully Treated with Follicular Unit Transplantation

Unger, Robin; Alsufyani, Mohammed A.

doi:10.1155/2011/129541

Cited by 11 publications

(5 citation statements)

References 7 publications

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“…Lezyon küçük ve lokalizeyse seçilmiş vakalarda cerrahi rezeksiyon yapılabilir (1). Bilateral TTA'sı olan bir olguda foliküler ünite transplantasyonu yapılmış, ve altı yıllık takipte saçlarda dökülme izlenmemiştir (9). Özellikle kozmetik kaygısı yüksek olan hastalara saç transplantasyonu önerilebilir.…”

Section: Olguunclassified

Two Cases of Temporal Triangular Alopecia

Hançer

2021

Muğla Sıtkı Koçman Üniversitesi Tıp Dergisi

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Temporal triangular alopesi (TTA), doğumda ya da erken çocukluk çağında ortaya çıkan, sıklıkla frontotemporal bölgede unilateral ya da bilateral yerleşim gösteren, asemptomatik, skar bırakmayan lokalize saç kaybıdır. Etkilenen bölgede tam bir saç kaybı olabilir ya da ince vellüs tüyleri görülebilir. Alopesi genellikle kalıcıdır. Çoğunlukla alopesi areata ile karıştırılmasından dolayı literatürde bildirilen TTA olguları az sayıdadır. Burada öykü ve klinik inceleme ile TTA tanısı alan iki olgu sunularak literatür gözden geçirilecektir.Temporal triangular alopecia (TTA) is an asymptomatic, nonscarring localized hair loss that occurs in birth or early childhood, often unilateral or bilateral in the frontotemporal region. The affected area may have a complete hair loss or thin vellus hairs. Alopecia is usually permanent. There are few cases of TTA reported in the literature because the majority of cases of TTA are misdiagnosed as alopecia areata. Here, two cases with TTA diagnosed by history and clinical examination will be presented and the literature will be reviewed.

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Section: Olguunclassified

Two Cases of Temporal Triangular Alopecia

Hançer

2021

Muğla Sıtkı Koçman Üniversitesi Tıp Dergisi

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“…Cutaneous and systemic manifestations are varied and may involve several organs and systems. Cutaneous: café au lait spots, nevus anemicus, hypochromic nevus, preauricular tags, plexiform neurofibroma, and linear morphea have been recorded in several studies, together with vitiligo, 44 congenital patches of alopecia, 41,55,56 multiple granular cell tumors, 35,57 hypoplastic nails, 35 unilateral lipohypoplasia, 14 and unilateral lymphedema. 38 Ocular: the most frequent findings were melanosis bulbi, glaucoma, iris mammillations, megalocornea, and buphthalmos.…”

Section: Associated Skin and Systemic Manifestationsmentioning

confidence: 99%

Phacomatosis Pigmentovascularis

et al. 2018

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The coexistence of a widespread vascular nevus and an extensive pigmentary nevus is defined as “phacomatosis pigmentovascularis” (PPV). More than 250 (sporadic) cases of PPV have been so far reported, mainly in Asian or Asian-related populations: mutations in genes related to angiogenic pathways (RAS, MAPK, mTOR, PI3K/AKT, and GNAQ) have been recently identified as the causes of this complex phenotype. In many cases, mutations in two different genes may coexist, representing the classical example of “twin spotting” phenomenon. PPV is usually associated with several extracutaneous anomalies including ocular manifestations (melanosis bulbi, glaucoma, iris mammillations, megalocornea, buphthalmos, strabismus, and hyperpigmentation of the conjunctiva, sclera, episclera, iris, trabecular meshwork, and choroid) and musculoskeletal alterations (limb hypertrophy, Klippel–Trenaunay type abnormalities, hemifacial hypertrophy, hemicorporal hypertrophy, macrocephaly, microcephaly, and scoliosis). Central nervous system anomalies have been reported in most of the patients and include seizure, cognitive delay, cerebral atrophy, hydrocephalus, sensorineural deafness, and intracranial hypertension, as well as migraine, pseudotumor cerebri, and intracerebral vascular malformations. More rarely, PPV has been associated with structural and/or vascular renal anomalies, hepatosplenomegaly, pyogenic granuloma, cavernous hemangioma, portal hypertension, umbilical hernia, hypoplasia of leg veins, and hypo- or hyperactivity of the immune system.

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“…This condition was first described in 1905 by Raymond Sabouraud in his book “ Manuel élémentaire de dermatologie topographique réégionale ” as “ alopecia triangulare congenitale de la temp .”[ 1 ] As of January 2015, 126 cases have been reported worldwide, as shown in Table 1 . [ 2 3 4 5 6 7 8 9 10 11 12 13 14 15 16 17 18 19 20 21 22 23 24 25 26 27 28 29 30 31 32 33 34 35 36 37 38 39 40 41 42 43 44 45 46 47 48 49 50 ] It typically affects the frontotemporal region [Figures 1 and 2 ], although there have been reported cases of CTA affecting the temporoparietal[ 15 42 46 ] and occipital scalp. [ 36 37 ] It is a nonprogressive disorder, which presents as a triangular, oval or lancet-shaped patch of alopecia.…”

Section: Introductionmentioning

confidence: 99%

“…Hair restoration surgery using follicular unit transplantation or surgical excision may provide patients with a satisfactory cosmetic result. [ 29 32 40 41 45 ] Long-term sustained benefits have been reported after 6 years of follow-up. [ 40 ]…”

Section: Introductionmentioning

confidence: 99%

Congenital triangular alopecia

Yesudian

2015

Int J Trichol

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Congenital triangular alopecia (CTA) also known as temporal triangular alopecia is a benign noncicatricial pattern of hair loss. It typically affects the frontotemporal region and rarely involves the temporoparietal or occipital scalp. It is a nonprogressive disorder that presents as a triangular, oval or lancet-shaped patch of alopecia. CTA can manifest at birth or develop later in life. The exact etiology of this condition remains unknown. Rarely, it may be associated with other disorders such as Down's syndrome and phakomatosis pigmentovascularis. The diagnosis is based on its distinct clinical appearance. Histologically, hair follicles are miniaturized and replaced by sparse vellus hair follicles. Tricoscopy using a polarized light handheld dermatoscope can be a useful diagnostic tool. CTA is often asymptomatic and remains unchanged throughout the life. No treatment is required. Surgical intervention with follicular unit hair transplantation can provide a satisfactory cosmetic result. In this paper, we have identified 126 cases of CTA in the published literature cited on PubMed between 1905 and 2015. From the available evidence, 79% of patients with CTA presented with unilateral hair loss, 18.5% with bilateral involvement and rarely, with occipital alopecia (2.5%). There was no gender predilection. These figures are entirely consistent with previously published data. Physicians should remember to consider CTA as a potential diagnosis in any patient presenting with a nonscarring alopecia in order to avoid unnecessary investigations and treatments.

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Bilateral Temporal Triangular Alopecia Associated with Phakomatosis Pigmentovascularis Type IV Successfully Treated with Follicular Unit Transplantation

Cited by 11 publications

References 7 publications

Two Cases of Temporal Triangular Alopecia

Two Cases of Temporal Triangular Alopecia

Phacomatosis Pigmentovascularis

Congenital triangular alopecia

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