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 Table of Contents  
CASE REPORT
Year : 2020  |  Volume : 12  |  Issue : 2  |  Page : 86-88  

Woolly hair nevus with trichotillomania in a 9-year-old boy: A rare case association


Department of Skin and V.D, Government Medical College, Kota, Rajasthan, India

Date of Submission05-Jan-2020
Date of Decision10-Jan-2020
Date of Acceptance07-Apr-2020
Date of Web Publication05-May-2020

Correspondence Address:
Devendra Yadav
KR-207, MBS Hospital Campus, Opposite to Circuit House, Nayapura, Kota, Rajasthan
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijt.ijt_103_19

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   Abstract 


Woolly hair nevus is a rare entity characterized by structural anomaly, which presents as curly hair in a circumscribed area of scalp, in a nonNegroid individual. These are abnormal hairs which are short, thin, and sparse. The term “woolly hair” refers to tightly coiled hair covering the whole scalp or part of it and has a hereditary character. Trichotillomania is a chronic impulse control disorder, characterized by the repetitive pulling out of one's own hair, leading to noticeable hair loss. Here, we present the case of a 9-year-old male child with woolly hair nevus over the occipital area, which developed at the age of 3 years; later, he developed trichotillomania at the age of 5 years, adjacent to the nevus. According to the best of our knowledge, there is no case report of woolly hair nevus presenting with trichotillomania till date.

Keywords: Curly hair, trichotillomania, woolly hairy nevus


How to cite this article:
Saini S, Kumar R, Yadav D. Woolly hair nevus with trichotillomania in a 9-year-old boy: A rare case association. Int J Trichol 2020;12:86-8

How to cite this URL:
Saini S, Kumar R, Yadav D. Woolly hair nevus with trichotillomania in a 9-year-old boy: A rare case association. Int J Trichol [serial online] 2020 [cited 2020 May 30];12:86-8. Available from: http://www.ijtrichology.com/text.asp?2020/12/2/86/283800




   Introduction Top


Woolly hair is an abnormal variant of the fine, curled hair on the scalp. Woolly hair nevus is a benign rare condition that is characterized by hair shaft abnormality, in which hairs become coiled and slightly hypopigmented.[1],[2] It usually develops in the first year of life, but can also appear in adolescence.[3] Both sexes are affected equally and may be associated with epidermal or melanocytic nevus. Trichotillomania is derived from the Greek “Thrix,” hair; “tillein,” pulling out; and “mania,” madness, which is one of the types of traumatic alopecia and accidental alopecia. It is recently grouped with obsessive-compulsive disorder in the DSM-5.


   Case Report Top


A 9-year-old male child presented to the dermatology outpatient department with his mother, complaining of a patch of curled and coiled hairs with altered texture over the occipital area of the scalp since he was 3 years old. She also gave a history of her child having fingers always on the scalp and plucking hairs near to that patch. Developmental milestones were normal, and there was no history of consanguinity in the family. The mother gave a history about him being avoided to go to school. On examination, there was a well-defined area of coiled, lighter, and altered texture hairs over the occipital area just below the vertex with no changes on the skin of the scalp. There was also an ill-defined patch with hairs of irregular length and broken hairs just adjacent to the woolly hair nevus [Figure 1]. Microscopy of the hair shaft was done, which showed that woolly hairs were thin, lighter in color comparative to normal hair, and there were angulation at some points [Figure 2]. Skin biopsy was not done as the diagnosis was clinical and also, the patient's mother did not give consent for biopsy. Trichoscopy of the adjacent patch was also done which showed uneven hair length, trichoptillosis, flame hair, coiled hair, follicular hemorrhage, bent hair, and “V” sign (Dermlite 3 dermoscope, ×10) [Figure 3].
Figure 1: A circumscribed area of rough and curly hairs with patch of trichotillomania over the occipital area of the scalp

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Figure 2: Microscopy of hair shows normal, thick, uniformly pigmented hair (yellow arrow); abnormal thin centrally hypopigmented woolly hair (green arrow); angulation at some points in the woolly hair (red arrow)

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Figure 3: Trichoscopy from the trichotillomanic patch shows angulated hairs (orange arrow); coiled hair (yellow arrow); flame hairs (green arrow); V-sign (purple arrow); tulip hairs (blue arrow); follicular hemorrhage (red arrow)

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Laboratory tests, electrocardiogram, abdominal ultrasound, and ophthalmological examination were normal. Based on all these findings, we arrived at a diagnosis of localized woolly hair nevus and trichotillomania.


   Discussion Top


The term “woolly hair” refers to tightly coiled hairs, which tend to be unruly, curly, lighter in color, and smaller in diameter than the surrounding normal hair without an increase in fragility and appear to be sparse.

Woolly hair nevus was first described by Wise in April, 1927.[4] Later, woolly hair was classified into three variants by Hutchinson et al.[5] as follows: autosomal dominant hereditary form which is generalized, autosomal recessive familial form, and nonhereditary localized form. The localized form is known as woolly hair nevus. Woolly hair nevus is further subdivided into three types: Type 1: without any skin and scalp changes, Type 2: associated with linear verrucous epidermal nevus, and Type 3: acquired progressive kinking of hair.[3] Woolly hair nevus appears in early childhood, as a circumscribed patch of kinky, very curly lighter hair, which is smaller in diameter than the surrounding normal hair without an increase in hair fragility and becomes less curly over the years.[6] Till date, there is no effective treatment for this condition, so parents are to be informed and counseled about the benign nature of the disease.

Trichotillomania, also known as hair-pulling disorder, keeps its place under obsessive-compulsive disorders. It is largely related to anxiety disorder with a lifetime prevalence of 0.6%.[7] Hair is pulled from anywhere on the body repeatedly, appearing as hair loss and marked function impairment.[7],[8] Triggers to pull may be multiple such as sensory, emotional, and cognitive. In many patients, phenomenon known as “automatic pulling;” “focused pulling” generally occur, in which patient is not fully aware of these pulling behaviors. It occurs when the patient sees or feels a hair that is not “right” or hair feels coarse, irregular, or “out of place.”[9] According to literature, dermoscopy shows decreased hair density, hair broken at different lengths, and short hair with trichoptilosis, which is diagnostic for trichotillomania.[10] There are several treatment options including cognitive and behavioral therapies, supportive counseling, and medications.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
   References Top

1.
Martín-González T, del Boz-González J, Vera-Casaño A. Woolly hair nevus associated with an ipsilateral linear epidermal nevus. Actas Dermosifiliogr 2007;98:198-201.  Back to cited text no. 1
    
2.
Torres T, Machado S, Selores M. Wooly hair generalizado: Case clinic e revisâo da literature. An Bras Dermatol 2010;85:97-100.  Back to cited text no. 2
    
3.
Post CF. Woolly hair nevus; report of a case. AMA Arch Derm 1958;78:488-9.  Back to cited text no. 3
    
4.
Wise F. Woolly hair nevus. A peculiar form of birthmark of the hair the scalp, hitherto undescribed, with report of two cases. Med J Rec 1927;125:545-7.  Back to cited text no. 4
    
5.
Hutchinson PE, Cairns RJ, Wells RS. Woolly hair. Clinical and general aspects. Trans St Johns Hosp Dermatol Soc 1974;60:160-77.  Back to cited text no. 5
    
6.
Usha V, Nair TV. Woolly hair nevus – Case report. Indian J Dermatol Venereol Leprol 1997;63:330-1.  Back to cited text no. 6
    
7.
Christenson GA, Pyle RL, Mitchell JE. Estimated lifetime prevalence of trichotillomania in college students. J Clin Psychiatry 1991;52:415-7.  Back to cited text no. 7
    
8.
Woods DW, Flessner CA, Franklin ME, Keuthen NJ, Goodwin RD, Stein DJ, et al. The trichotillomania impact project (TIP): Exploring phenomenology, functional impairment, and treatment utilization. J Clin Psychiatry 2006;67:1877-88.  Back to cited text no. 8
    
9.
Christenson GA, Mansueto CS. Trichotillomania: Descriptive characteristics and Phenomenology. In: Trichotillomania. Stein DJ, Christianson GA, Hollander E, editors. Washington, DC: American Psychiatric Press; 1999. p. 1-41.  Back to cited text no. 9
    
10.
Shim WH, Jwa SW, Song M, Kim HS, Ko HC, Kim BS, et al. Dermoscopic approach to a small round to oval hairless patch on the scalp. Ann Dermatol 2014;26:214-20.  Back to cited text no. 10
    


    Figures

  [Figure 1], [Figure 2], [Figure 3]



 

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