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CONCISE COMMUNICATION
Year : 2020  |  Volume : 11  |  Issue : 2  |  Page : 263-265  

A case of familial trichomegaly with synophrys in association with loose anagen syndrome


1 Department of Dermatology, Venereology and Leprosy, Dr. Ram Manohar Lohia and Post Graduate Institute of Medical Education and Research, New Delhi, India
2 Department of Pathology, Dr. Ram Manohar Lohia and Post Graduate Institute of Medical Education and Research, New Delhi, India

Date of Web Publication9-Mar-2020

Correspondence Address:
Surabhi Sinha
Department of Dermatology, Venereology and Leprosy, Dr. Ram Manohar Lohia and Post Graduate Institute of Medical Education and Research, New Delhi
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/idoj.IDOJ_16_19

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How to cite this article:
Mathachan SR, Sinha S, Sardana K, Malhotra P. A case of familial trichomegaly with synophrys in association with loose anagen syndrome. Indian Dermatol Online J 2020;11:263-5

How to cite this URL:
Mathachan SR, Sinha S, Sardana K, Malhotra P. A case of familial trichomegaly with synophrys in association with loose anagen syndrome. Indian Dermatol Online J [serial online] 2020 [cited 2020 Mar 28];11:263-5. Available from: http://www.idoj.in/text.asp?2020/11/2/263/261219



Sir,

A 17-year-old Indian girl presented to our department with complaints of slow growing, sparse, dry and light colored scalp hair with history of diffuse and excessive shedding of hair since birth. She was incidentally detected to have abnormally long and curly eyelashes measuring 18 mm in the center [Figure 1], 13 mm in the peripheral edge [Figure 2], and thick eyebrows with synophrys [Figure 3]. Scalp hair was short, dry, thin, hypopigmented, and of uneven length with decreased hair density [Figure 4] and [Figure 5]. Her 28-year-old elder brother [Figure 6] and 11-year-old younger brother [Figure 7] were revealed to have abnormally long eyelashes and synophrys too, albeit slightly mild compared with their sister. A family tree did not reveal any other affected family member. History of fourth degree consanguineous marriage of their parents [ first cousins] was noted [Figure 8]. Systemic, dental, physical, or mental abnormalities and drug intake were ruled out. Hair pull test was positive with easy and painless extraction of anagen hairs. Hair trichogram showed anagen hairs (<70%) with misshapen hair bulbs and absent root sheaths. Electron microscopy of hair could not be performed due to non-availability at our center. Routine biochemical and endocrinological tests of the girl were normal.
Figure 1: Eyelash measurement at the center

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Figure 2: Eyelash measurement at peripheral edge

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Figure 3: 17 year old girl with eyelash Trichomegaly and synophrys (sister)

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Figure 4: Short, dry, thin and hypopigmented hair; Loose anagen syndrome

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Figure 5: Lateral view of the scalp showing loose anagen hair

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Figure 6: Eyelash trichomegaly in elder brother

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Figure 7: Eyelash trichomegaly in younger brother with synophrys

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Figure 8: Pedigree of the family with trichomegaly. Black squares (men) and circles (women) indicate affected persons; white squares and circles indicate unaffected persons; double lines, consanguineous marriages; single lines, nonconsanguineous marriages; and arrow, the index case

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Eyelash trichomegaly is an unusual disorder characterized by an increase in the length, pigmentation, curling or thickness of eyelashes with more than 12 mm long eyelashes in the central area or more than 8 mm in the peripheral region.[1] It was first reported by Harrison et al. in 1997 in an 18 month old female child and her siblings.[2] It may be inherited either as part of congenital syndromes or as an isolated trait or it may be associated with an acquired illness or drug intake. Two congenital syndromes with trichomegaly as their defining distinguishing feature are Cornelia de Lange syndrome and Oliver McFarlane syndrome.[1] Eyelash trichomegaly has been reported with several diseases including human immunodeficiency virus infection, atopic dermatitis, alopecia areata, systemic lupus erythematosus, dermatomyositis, allergic rhinitis and uveitis. In addition, treatment with topical prostaglandins, cyclosporine, tacrolimus, topiramate, zidovudine and epidermal growth factor receptor inhibitors can cause trichomegaly as a side effect.[3] No history suggestive of any congenital syndrome, acquired diseases or drug intake were present in our patient.

Loose anagen hair syndrome (LAHS) is a disorder in which hair shaft is loosely anchored to the follicle resulting in painless easily pluckable hairs which are devoid of root sheaths. The three most important clinical signs of LAHS are reduced hair length, altered hair texture and increased hair shedding.[4] The management of LAHS is conservative and most cases resolve spontaneously with age. Topical minoxidil can be used as a first line medication in severe cases.[5] To our knowledge, familial trichomegaly in association with LAHS is a hitherto unreported association. Thus we are reporting this case of familial trichomegaly with synophrys in three siblings with coexistent LAHS in one of them, due to its rarity.

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.
Dalal A, Sharma S, Kumar A, Sharma N. Eyelash trichomegaly: A rare presenting feature of systemic lupus erythematosus. Int J Trichology 2017;9:79-81.  Back to cited text no. 1
    
2.
Harrison DA, Mullaney PB. Familial trichomegaly. Arch Ophthalmol 1997;115:1602-3.  Back to cited text no. 2
    
3.
Tamer F, Yuksel ME. Familial eyelash trichomegaly: The case of a sister and a brother. Indian J Dermatol Venereol Leprol 2016;82:700-2.  Back to cited text no. 3
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4.
Tosti A, Piraccini BM. Loose anagen hair syndrome and loose anagen hair. Arch Dermatol 2002;138:521-2.  Back to cited text no. 4
    
5.
Cantatore Francis JL, Orlow SJ. Practical guidelines for evaluation of loose anagen hair syndrome. Arch Dermatol 2009;145:1123-8.  Back to cited text no. 5
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6], [Figure 7], [Figure 8]



 

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