DR [Dermatology Reports 2011; 3:e35] [page 75] Unilateral Blaschkoid lichen planus in successive pregnancies Shiva Kumar, Rajendra Okade, Yasmin Abdul Rahman Department of Dermatology, Venereology and Leprosy, Sri DevarajUrs, Medical College, Tamaka, Kolar, India Abstract A number of genetic, congenital and acquired dermatoses have been known to fol- low Blaschko’s lines. A common disorder like lichen planus can very rarely present with pruritic lesions in atypical patterns such as unilateral distribution, painful eruptions and along Blaschko’s lines. Various triggering fac- tors varying from viral infections and vaccina- tions to trauma have been implicated in lichen planus. We describe a female patient in the second trimester of her second preg- nancy who developed unilateral lichen planus along Blaschko’s lines during both pregnan- cies. No case of lichen planus along Blaschko’s lines recurring during pregnancy is reported so far. Could pregnancy itself be a contributory factor towards onset of lichen planus in this case? Introduction Lichen planus (LP) is a common inflamma- tory disorder that affects skin, mucous mem- brane, nails and hair. It is classically charac- terised by pruritic, shiny, violaceous, flat topped, polygonal papules which retain skin lines and maybe closely aggregated or widely dispersed.1 Various etiological factors include immunological mediation, chronic liver dis- ease, hepatitis C infection2-3 and hereditary causes among others . Variants of this disor- der include hypertrophic LP, follicular LP, lin- ear LP, annular LP, vesicobullous LP, actinic LP, erosive and nonerosive mucous mem- brane LP.1 Blaschko’s lines were delineated in 1901 by a German dermatologist Alfred Blaschko . The original description by Blaschko referred to a system of lines on the human skin which the linear naevi and dermatoses follow.4 The pat- tern is attributed to lines of migration of epi- dermal cells during embrogenesis.5 These lines are curved over scalp and face, V - shaped in upper spine, inverted U shaped over chest and upper arm, S – shaped whorls on the chest and abdomen and perpendicular over the front and back of extremities. Types of Blaschko’s pattern include narrow band, large band, checkerboard and phylloid.6 Case Report A 24-year-old female in the 2nd trimester of her 2nd pregnancy presented with a history of itchy skin lesions which started over dorsum of her left hand 3 months back. New lesions progressively appeared in a linear pattern extending proximally to involve the left fore- arm, arm and shoulder. Simultaneously new elevated discoloured lesions appeared over left flank, abdomen and breast. Patient had similar lesions in the same areas during her 1st pregnancy 2 year’s back which had spontaneously resolved within 3 months after delivery with residual post inflammatory hyperpigmentation in the affected areas which still persisted. On examination, violaceus flat topped 2-5 mm papules were noted, some of which showed typical lacy white pattern of Wickham’s striae extending from left thumb uptoleft arm in a wave like pattern along Blaschko’s lines (Figure 1), they were dis- crete at few places while at others they appeared to be coalescing. Similar papules in a wavy pattern were noted over left shoulder, upper back (Figure 2), discontinuous papules and hyperpigmented macules were seen over left flank, abdomen (Figure 3) and left breast in 3 curved lines. Post inflammatory hyperpig- mented macules of lesions which developed during the previous pregnancy were seen along the same Blaschko’s lines. None of the eruptions crossed the midline. The patient had no oral lesions or nail changes. A working diagnosis of lichen planus was made. Preliminary routine investigations were with- in normal limits and tests for Hepatitis B and C were negative. Punch biopsy of left forearm lesions showed typical features of lichen planus (Figure 4). Discussion Lichen planus is a subacute to chronic der- matosis, which is benign and self limiting , although recurrences can occur. The disease has a predilection for flexor surfaces of fore- arms, legs and glans penis. Inverse lichen planus eruption occurs in flexures like axilla, inframammary folds and groins. Eruptions may be localised or extensive and Koebner’s phenomenon is commonly seen. Oral lesions of lichen planus are frequently seen either as sole manifestations or associated with cuta- neous involvement. Apart from the known variants of lichen planus, a few rare cases of lichen planus along Blaschko’s lines has been described.1 Histopathology examination reveals com- pact orthokeratosis, wedge shaped hypergran- ulosis irregular acanthosis, vacuolar alter- ation of the basal layer. Rete ridges may show saw - tooth appearance. Degenerating epider- mal cells form colloid bodies and a band like infiltration of lymphocytes may obliterate the dermo - epidermal junction.7 Blaschko’s lines do not correspond to any vascular, lymphatic or neural structures and are invisible and become apparent in certain disease states such as pigmentary disorders (naevus achromicus, epidermal naevus), X linked genetic skin disease (Incontinentia pigmenti) , acquired inflammatory skin disor- ders (LP, lichen striatus) and chimerism.6 Case reports of unilateral cutaneous lichen planus,1,8-9 lichen planus along with unilateral mucosal involvement,10 linear lichen pigmen- tosus11 and painful atrophic lichen planus12 have been reported along Blaschko’s lines. Long et al. described a case of bilateral linear lichen planus along Blaschko’s lines.13 Wolfs isotopic response is the occurrence of new skin disorder exactly at the site of another, unrelated and already resolved skin disease, this phenomenon has been seen in der- matomal lichen planus following herpes zoster at the same location.14 Krasowska et al. reported a case of a 33 year old healthy woman who developed recurrent unilateral lichen planus along Blaschko’s lines following 3 successive deliveries of healthy babies15. Our patient presented with history of onset of lesions during first trimester and to the best of our knowledge this is the first case of lichen planus along Blaschko’s lines occur- ring during successive pregnancies. Considering the fact that various known and idiopathic causes have been known to trigger Dermatology Reports 2011; volume 3:e35 Correspondence: Shiva Kumar, No. 66, 11th cross, Indiranagar, 1st stage, Bangalore- 560038, India. Tel. +91-9886151969. E-mail: dermashiva@rediffmail.com Key words: lichen planus, pregnancy, Blaschko’slines. Received for publication: 23 August 2011. Accepted for publication: 6 September 2011. This work is licensed under a Creative Commons Attribution NonCommercial 3.0 License (CC BY- NC 3.0). ©Copyright S. Kumar et al., 2011 Licensee PAGEPress, Italy Dermatology Reports 2011; 3:e35 doi:10.4081/dr.2011.e35 No n- co mm er cia l u se on ly [page 76] [Dermatology Reports 2011; 3:e35] lichen planus, a need to follow up the patient in current and subsequent pregnancies becomes essential to find a positive correla- tion between pregnancy and lichen planus and to establish whether pregnancy itself could have triggered lichen planus. References 1. Lehman JS, Tollefson MM, Gibson LE. Lichen planus. Int J Dermatol 2009;48:682- 94. 2. Bellman B, Reddy RK, Falanga V. Lichen planus associated with hepatitis C. Lancet 1995;346:1234. 3. Cribier B, Garnier C, Laustriat D, Heid E. Lichen planus and Hepatitis C virus infec- tion: an epidemiologic study. J Am Acad Dermatol 1994;31:1070-2. 4. Harper JI ,Trembath RC. Genetics and gen- odermatosis. In: Burns T, Breathnach S, Cox N, Griffith C, editors. Rook’s Text book of Dermatology. 7th ed . Oxford: Blackwell Science; 2004. pp. 12-17. 5. McGrath JA, Mclean WHI. Genetics in rela- tion to skin. In: Wolff K , Goldsmith LA , Stephen IK, et al., editors. Fitzpatrick’s Dermatology in general medicine. 7th ed. New York, NY: McGraw -Hill; 2008. p. 83. 6. Bolognia JL, Orlow SJ, Glick SA. Lines of Blaschko. J Am Acad Dermatol 1994; 31:157- 90. 7. Mobini N, Toussaint S, Kamino H. Noninfectious erythematous, papular and squamous diseases. In: Elder DE, editor . Lever’s histopathology of the skin. 10th ed. Philadelphia, PA: Lippincott Williams and Wilkins; 2010. pp.185-186. 8. Saxena AK, Nigam PK. Unilateral Lichen planus. Cutis 1988;42:142-3. 9. Gupta AK, Gorsulowsky DC. Unilateral lichen planus: An unusual presentation. Arch Dermatol 1987;123:295-6. 10. Hartl C, Steen KH, Wegner H, et al. Unilateral linear lichen planus with mucous membrane involvement. Acta DermVenerol 1999;79:145-6. 11. Seo JK, Lee HJ, Lee D, et al. A case of lin- ear lichen planus pigmentosus. Ann Dermatol 2010;22:323-5. 12. Lakshmi C, Divakaran J, Sivaraman A, et al. Painful linear atrophic lichen planus along lines of Blaschko. Ind J Dermatol 2006;51:42-3. 13. Long CC, Finlay AY. Multiple linear lichen planus in the lines of Blaschko. Br J Dermatol 1996;135:275-6. 14. Braun RP, Barua D, Masouye I. Zosteriform lichen plaus after herpes zoster. Dermatology 1998;197:87-8. 15. Krasowska D, Pietrzak A, Lecewicz-Torun B. Unilateral multiple linear lichen planus following the Blaschko lines recurring after deliveries. Dermatology 2001;202: 340. Case Report Figure 1. Typical violaceous papules over thumb and forearm in a linear distribution. Figure 3. Gravid abdomen showing linear pigmented macules and papules. Figure 4. Histopathology showing dense lichenoid infiltrate and saw toothed rete ridges. Figure 2. Pigmented papules and macules over shoulder. No n- co mm er cia l u se on ly