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E-IJD® - CORRESPONDENCE |
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| Year : 2022 | Volume
: 67
| Issue : 1 | Page : 94 |
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| Atypical localization of congenital triangular alopecia associated with Down's syndrome |
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Kanade Shimada1, Ryota Hayashi1, Rei Yokoyama1, Osamu Ansai1, Satoru Shinkuma1, Yutaka Shimomura2, Riichiro Abe1
1 Division of Dermatology, Niigata University Graduate School of Medical and Dental Sciences, Niigata, Japan
2 Department of Dermatology, Yamaguchi University Graduate School of Medicine, Ube, Yamaguchi, Japan
| Date of Web Publication | 19-Apr-2022 |
Correspondence Address:
Ryota Hayashi
Division of Dermatology, Niigata University Graduate School of Medical and Dental Sciences, Niigata
Japan
 Source of Support: None, Conflict of Interest: None  | Check |
DOI: 10.4103/ijd.ijd_1026_20
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How to cite this article:
Shimada K, Hayashi R, Yokoyama R, Ansai O, Shinkuma S, Shimomura Y, Abe R. Atypical localization of congenital triangular alopecia associated with Down's syndrome. Indian J Dermatol 2022;67:94 |
How to cite this URL:
Shimada K, Hayashi R, Yokoyama R, Ansai O, Shinkuma S, Shimomura Y, Abe R. Atypical localization of congenital triangular alopecia associated with Down's syndrome. Indian J Dermatol [serial online] 2022 [cited 2023 Jun 4];67:94. Available from: https://www.e-ijd.org/text.asp?2022/67/1/94/343286 |
Sir,
Congenital triangular alopecia (CTA), also known as temporal triangular alopecia, is a circumscribed, non-cicatricial, and non-inflammatory type of alopecia.[1] CTA is rare and typically presents in the first 10 years of life. To date, several cases of CTA with congenital disorders, including Down's syndrome (DS), have been described.[2],[3] Here, we report a case of atypical localization of CTA in a patient with DS.
A 4-year-old boy with DS was referred to our hospital for non-scarring alopecia that had been present since birth. He had been diagnosed with alopecia areata (AA) which did not improve with topical application of corticosteroids. A triangular-shaped, non-scarring, and non-inflammatory patch with vellus hair was observed on his left parietal scalp [Figure 1]. Trichoscopy findings showed white hairs, vellus hair surrounded by terminal hair, and empty follicles [Figure 2]. A skin biopsy was not performed because the patient's parents did not provide consent for it. | Figure 1: Clinical findings (a) A triangular-shaped, non-scarring, and non-inflammatory patch is seen on his parietal scalp. (b) White hair (blue arrow) and vellus hair (red arrow) in the lesion
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 | Figure 2: Trichoscopy findings (a) White hair (blue arrow) and vellus hair (red arrow) in the lesion. (b) White hairs (blue arrow), vellus hair surrounded by terminal hair (red arrow), and empty follicles (red circle) are visible on trichoscopy
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We considered the following differential diagnoses: AA, aplasia cutis congenita, and traumatic alopecia. However, the absence of exclamation mark hair, scarring alopecia, and birth trauma led us to exclude these disorders. Although the localization was atypical for CTA in our patient, trichoscopy findings were consistent, and a diagnosis of CTA was made.
CTA is a rare hair disease, and the patients with CTA are basically diagnosed based on clinical findings. To diagnose CTA, we need to exclude several diseases, including AA and aplasia cutis. Trichoscopic features typical of CTA such as white hairs, vellus hairs surrounded by terminal hairs, and empty follicles are crucial for diagnosis.[4] Although in many CTA cases, the temporal areas are triangular, several cases of CTA occur in atypical areas and shapes of it are not a triangle.[4] We suggested the following criteria for diagnosis of CTA: (1) triangular or spear-shaped hair loss involving the frontotemporal scalp; (2) dermoscopy revealing normal follicle openings with vellus hairs surrounded by normal terminal hairs without yellow and black dots, dystrophic hairs, or decreased follicular openings; and (3) no significant hair regrowth after confirmation of the presence of vellus hairs.
DS is the most common chromosomal abnormality and is associated with various skin lesions such as AA. The frequency of AA is higher in those with DS than in normal individuals. A previous report demonstrated that 6.9% of patients with DS have AA.[5] To the best of our knowledge, only one case of CTA associated with DS has been reported.[2],[3] Although in most cases, CTA occurred in the temporal area, a different localization was noted in our case. The difference of area was not related to DS.[2],[3] Several cases of CTA are familial, suggesting that a causative gene may exist. However, it has not been identified.[2] Although there is no consistently effective treatment for CTA, some previous reports showed that hair transplantation and excision surgery were successful.[3]
We reported a case of CTA associated with DS. Although CTA is frequently misdiagnosed, this case highlights that trichoscopic signs are crucial to exclude other diseases and diagnose CTA.
Statement on consent for publication
The patient and patient's parents gave their full permission for the publication, reproduction or other use of photographs.
Financial support and sponsorship
Nil.
Conflicts of interest
There are no conflicts of interest.
 References | |  |
| 1. | Tosti A. Congenital triangular alopecia. Report of fourteen cases. J Am Acad Dermatol 1987;16:991-3.  |
| 2. | Yamazaki M, Irisawa R, Tsuboi R. Temporal triangular alopecia and a review of 52 past cases. J Dermatol 2010;37:360-2. |
| 3. | Bordel-Gómez MT. Congenital triangular alopecia associated with Down's syndrome. J Eur Acad Dermatol Venereol 2008;22:1506-7. |
| 4. | Fernández-Crehuet P, Vaño-Galván S, Martorell-Calatayud A, Arias-Santiago S, Grimalt R, Camacho-Martínez FM. Clinical and trichoscopic characteristics of temporal triangular alopecia: A multicenter study. J Am Acad Dermatol 2016;75:634-7. |
| 5. | Rork JF, McCormack L, Lal K, Wiss K, Belazarian L. Dermatologic conditions in Down syndrome: A single-center retrospective chart review. Pediatr Dermatol 2020. doi: 10.1111/pde.14214. |
[Figure 1], [Figure 2] |
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