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CASE REPORT
Year : 2018  |  Volume : 19  |  Issue : 4  |  Page : 354-356

Lymphoplasmacytic plaque in children: An emerging new clinical entity


1 Department of DVL, GSL Medical College, Rajahmundry, Andhra Pradesh, India
2 Dermatopathologist, London Dermatopathology Group (LD Path Group), Andhra Pradesh, India

Date of Web Publication28-Sep-2018

Correspondence Address:
Dr. Raghurama Rao Gandikota
Department of DVL, GSL Medical College, Rajanagaram, Rajahmundry - 533 296, Andhra Pradesh
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijpd.IJPD_115_17

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  Abstract 


Pretibial lymphoplasmacytic plaque in children is a rare emerging clinicopathological entity characterized by asymptomatic benign solitary plaque. Most of the cases are reported in healthy females. The treatment of choice is topical steroids.

Keywords: Children, dermal lymphoplasmacytic infiltration, female, topical steroids


How to cite this article:
Ganni S, Attili SK, Gandikota RR, Haritha K. Lymphoplasmacytic plaque in children: An emerging new clinical entity. Indian J Paediatr Dermatol 2018;19:354-6

How to cite this URL:
Ganni S, Attili SK, Gandikota RR, Haritha K. Lymphoplasmacytic plaque in children: An emerging new clinical entity. Indian J Paediatr Dermatol [serial online] 2018 [cited 2020 Oct 25];19:354-6. Available from: https://www.ijpd.in/text.asp?2018/19/4/354/242407




  Introduction Top


Lymphoplasmacytic plaque (LPP) in children is an emerging condition whose clinicopathological characterization continues to evolve. It was originally termed “isolated benign primary cutaneous plasmacytosis in children” by Gilliam.[1] Fried et al. considered the term “pretibial lymphoplasmacytic plaque in children” more adequate for describing this more elusive disease.[2] This clinical condition is characterized by asymptomatic benign erythematous plaque over the tibia with dermal lymphoplasmacytic infiltrate. Till date, nine cases have been reported typically in healthy Caucasian and Asian children.[3] Herein, we report the case of an 11-year-old healthy female child with pretibial plaque.


  Case Report Top


An 11-year-old healthy girl presented with a single asymptomatic erythematous plaque on the shin of the right leg with 9 years' duration. Her mother observed small papular lesions at the site when the child was 2 years old and the lesions were gradually increasing to form a plaque of the present size. She had no history of trauma, insect bite, or foreign body exposure. On examination, there was a well-defined, nontender, erythematous plaque of 2 cm × 2 cm diameter over the anterior aspect of the right leg [Figure 1]. There were no similar lesions elsewhere over the body. There was no regional lymphadenopathy. Systemic examination was normal. All routine hematological and biochemical investigations were within normal limits. Mantoux test was negative. Serological tests for syphilis, HIV, and HbsAg were negative. A provisional diagnosis of cutaneous tuberculosis, insect bite reaction, and regressing hemangioma was considered. Four-millimeter punch biopsy of the skin specimen showed epidermis with mild atrophy and prominent lichenoid interface dermatitis with numerous macrophages engulfing colloid bodies. Within the dermis, there was a top heavy superficial and deep confluent perivascular inflammatory infiltrate composed of lymphocytes, plasma cells, and few histiocytes. The infiltrate extends into the fat (lobular panniculitis) [Figure 2], [Figure 3], [Figure 4]. No clonality or atypicality of lymphocytes was present. Special stains such as periodic acid–Schiff, Fite, Leishman, Gomori-methenamine silver were done to rule out fungal, mycobacterial, and parasitic infections. A diagnosis of LPP was made on the basis of clinical and histopathological features. A potent topical steroid (clobetasol propionate 0.1%) cream was prescribed, and the case was reviewed after 8 weeks. There was a good response with three-fourths of the plaque regressed and the topical steroid was continued for another 4 weeks and the lesions completely regressed. The case was followed up to 6 months and there was no recurrence.
Figure 1: Erythematous plaque of 2 cm × 2 cm on the shin

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Figure 2: Low-power image showing a superficial and deep dense top heavy infiltrate extending into superficial panniculus (H and E, ×20)

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Figure 3: Dense confluent lymphoplasmacytic lichenoid infiltrate with epidermal atrophy, colloid bodies, and melanophages (H and E, ×400)

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Figure 4: Dense lymphoplasmacytic infiltrate extending in and around the fat lobules (H and E, ×400)

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  Discussion Top


LPP in children is characterized by asymptomatic solitary pink-to-violaceous erythematous papulonodular plaque with diameter measuring a few centimeters with a variably smooth-to-lobulated surface and mild scaling. The age of the affected individuals varies from 7 to 15 years with a predilection for females. The anterior shins appear to be the common sites, but lesions can also occur on the buttocks. Histopathologically, LPP shows a dense, mixed infiltrate featuring lymphocytes with admixed plasma cells. An epithelioid granulomatous infiltrate can also be seen.[3] Till date, nine cases have been reported and ours is the tenth case. The details of these cases are summarized in [Table 1]. Clinically pretibial LPP must be differentiated from the following conditions: infectious diseases, histiocytic tumors, vascular lesions, spitzoid proliferation, and lichenoid conditions. The condition is not associated with any systemic disease.[6]
Table 1: Reported cases of lymphoplasmacytic plaque in children

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A wide variety of histopathological conditions such as cutaneous plasmacytoma, primary cutaneous plasmacytosis, and end-stage multiple myeloma must be considered as the differential diagnosis. In contrast to this group of disorders, LPP in children does not feature lymphoid or plasmacytic atypia and clonality.[6] The main differential diagnosis is with other reactive infiltrates including acral pseudolymphomatous angiokeratoma of children (APACHE), but this condition is characterized by multiple, unilateral persistent angiomatous papules or nodules on the acral regions of hands and feet.[5] In APACHE, histologically, the nodules are characterized by well-circumscribed nodular dermal infiltrate composed of lymphocytes, histiocytes, plasma cells, and thick-walled long blood vessels lined with plump endothelial cells.[5] Our case is strikingly similar clinically and histopathologically with the published nine cases and fulfills the criteria to say that it is a case of pretibial LPP.

The exact pathogenesis of this benign condition is not known, but may be the lesions represent an unusual local reactive process to yet unknown antigen.[2] LPP is a benign chronic condition; although there was no effective treatment, the plaques can be treated for cosmetic reasons with potent local steroids or intralesional steroid injections with varying outcomes. Pulsed dye laser induced a certain degree of fading of purplish color. Complete excision of the plaque is the final answer.[5]

Acknowledgments

We thank and acknowledge Dr. Alistain Robson for his valuable suggestions toward establishing at this diagnosis.

Consent

Patient's declaration and consent form was obtained.

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.
Gilliam AC, Mullen RH, Oviedo G, Bhatnagar R, Smith MK, Patton DF, et al. Isolated benign primary cutaneous plasmacytosis in children: Two illustrative cases. Arch Dermatol 2009;145:299-302.  Back to cited text no. 1
    
2.
Fried I, Wiesner T, Cerroni L. Pretibial lymphoplasmacytic plaque in children. Arch Dermatol 2010;146:95-6.  Back to cited text no. 2
    
3.
Seo J, Kim JY, Kim SH, Oh SH. Lymphoplasmacytic plaque in children. Ann Dermatol 2016;28:266-8.  Back to cited text no. 3
    
4.
Ahn JJ, Yang YS, Shin MK, Lee SW, Kim NI. Case of isolated benign primary cutaneous plasmacytosis in a child. J Dermatol 2011;38:364-7.  Back to cited text no. 4
    
5.
Moulonguet I, Hadj-Rabia S, Gounod N, Bodemer C, Fraitag S. Tibial lymphoplasmacytic plaque: A new, illustrative case of a recently and poorly recognized benign lesion in children. Dermatology 2012;225:27-30.  Back to cited text no. 5
    
6.
Porto DA, Sutton S, Wilson JB, Scupham RK, Stone MS, Liu V, et al. Lymphoplasmacytic plaque in children: A report of two new cases with review of the literature. J Cutan Pathol 2013;40:50-5.  Back to cited text no. 6
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4]
 
 
    Tables

  [Table 1]



 

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