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DERMATOPATHOLOGY ROUND
Year : 2013  |  Volume : 58  |  Issue : 4  |  Page : 278-280
Pseudolymphomatous folliculitis: A distinctive cutaneous lymphoid hyperplasia


Department of Dermatology, Dr. L. H. Hiranandani Hospital, Powai, Mumbai, India

Date of Web Publication25-Jun-2013

Correspondence Address:
Rameshwar M Gutte
Department of Dermatology, OPD No. 112, 1st Floor, Dr. L. H. Hiranandani Hospital, Powai, Mumbai - 400 076
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0019-5154.113937

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   Abstract 

Pseudolymphomatous folliculitis (PLF) was first described in 1986 as a distinct variant of pseudolymphoma, characterized by a dense lymphoid infiltrate and accompanied by hyperplastic hair follicles. Here in we report a case of PLF presenting as an erythematous plaque with pustules and satellite lesions on forehead in an otherwise healthy adult male patient.


Keywords: Forehead, lymphoma, pseudolymphomatous folliculitis


How to cite this article:
Gutte RM. Pseudolymphomatous folliculitis: A distinctive cutaneous lymphoid hyperplasia. Indian J Dermatol 2013;58:278-80

How to cite this URL:
Gutte RM. Pseudolymphomatous folliculitis: A distinctive cutaneous lymphoid hyperplasia. Indian J Dermatol [serial online] 2013 [cited 2019 Aug 21];58:278-80. Available from: http://www.e-ijd.org/text.asp?2013/58/4/278/113937

What was known? Pseudolymphomatous folliculitis is a poorly defined entity among the cutaneous pseudolymphomas.



   Introduction Top


Pseudolymphomatous folliculitis (PLF) is a poorly defined entity among the cutaneous pseudolymphomas. Clinically, PLF is typified by a solitary dome-shaped or flat-elevated nodule, located on the face, scalp, and trunk. However, multiple lesions may occur. [1],[2] It is sometimes difficult to differentiate from primary malignant cutaneous lymphomas that may present as solitary lesions. [2] PLF is unique in the clinical appearance and histopathological features compared with other cutaneous pseudolymphomas. [3]


   Case Report Top


A 45-year-old married male presented to us with a red raised lesion on the right side of forehead. The lesion has started 2 months back as single papule and gradually progressed to present size. Recently, he also noticed development of new papules around it and some pustules on it [Figure 1]. There was no history of any drug intake, insect bite or any local trauma prior to lesions. Before presenting to us, patient was treated with various topical and oral antibiotics like amoxicillin-clavulanic acid combination, azithromycin and linezolid etc., but lesions kept on increasing without any improvement.
Figure 1: An erythematous infiltrated plaque with pustules and surrounding erythematous papules on forehead

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Cutaneous examination erythematous indurated minimally tender plaque of 2 cm × 4 cm on the right side of the forehead topped with few pustules and brownish crust at places. Furthermore, there were two discrete erythematous papules of recent onset developing around it. There was no any lymph node swelling noted. Rest of his cutaneous and systemic examination was unremarkable. Ophthalmic examination revealed no abnormality.

A diagnosis of fixed cutaneous leishmaniasis, deep fungal infection and pseudolymphoma was thought. Pus swab from lesion showed no growth. A biopsy of the lesion revealed dense lymphocytic infiltrate extending from upper dermis to lower dermis [Figure 2]a. Involvement of subcutis was not seen. Infiltrate also contained numerous histiocytes, few neutrophils and occasional plasma cells that surrounded and infiltrated hypertrophic hair follicles with dilated sebaceous glands (so called activated pilosebaceous units). Some lymphocytes infiltrated into the hair follicles leading to its destruction and ultimately effacement by infiltrate [Figure 2]b. Infiltrated cells were medium to large sized lymphocytes with hyperchromatic nuclei, high nuclear/cytoplasmic ratio, and prominent nucleoli accompanied by numerous histiocytes, netrophils, and few plasma cells [Figure 3]. No any bacterial or fungal elements were identified. On clinicopathological correlation a diagnosis of PLF was made. Due to economic constraints, immunohistochemistry for T- and B-cell markers or cluster designation 1a could not be done. Patient was advised topical clobetasol and fusidic acid combination in a cream form. Also oral prednisolone 40 mg per day with antacids and oral calcium supplement which was tapered over 40 days. Intra-lesional triamcinolone injection (20 mg/ml) was given after 7 days of starting oral steroids. Remarkable improvement was seen after 4 days of single injection [Figure 4].
Figure 2: (a) Dense lymphocytic dermal infiltrate accompanied by hyper-plastic follicles and dilated activated pilosebaceous unit (H and E, ×40). (b) Atypical lymphocytic infiltrate invading hair follicle with destruction of it (H and E, ×100)

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Figure 3: High power view showing large, atypical, hyperchromatic lymphocytes with increased nucleo-cytoplasmic ratio along with histiocytes and few neutrophils (H and E, ×400)

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Figure 4: Remarkable improvement after oral prednisolone and single intra-lesional steroid injection

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   Discussion Top


Pseudolymphomatous folliculitis is a poorly defined entity among the cutaneous pseudolymphomas. Clinically, PLF is typified by a solitary dome-shaped or flat-elevated nodule, located on the face, scalp and trunk. However, multiple lesions may occur. [1],[2] It is sometimes difficult to differentiate from primary malignant cutaneous lymphomas that may present as solitary lesions. [2] PLF is unique in the clinical appearance and histopathological features compared with other cutaneous pseudolymphomas. [3]

The histological criteria for diagnosis includes dense and nodular dermal lymphocytic infiltrates from the dermis to the subcutis, befitting the term pseudolymphoma, in which lymphocytes surround, infiltrate pilosebaceous unit and deform the walls. [3],[4] Two types of follicular invasion can be recognized: In one type, hair follicles are destroyed and ultimately effaced, whereas in the other type, the hair follicles are not damaged but activated and show hyperplastic changes with various irregularly enlarged shape of the follicular wall. [4],[5] Our case had both, the activated pilosebaceous units with dilated, enlarged, irregularly shaped follicular walls and infiltrate invading the follicle with destruction of it. Along with mixed population of CD79a positive B-cells and CD3, CD4, CD8 positive T-cells, increased numbers of perifollicular histiocytes expressing anti-S-100 protein and CD1a is reported as characteristic feature of PLF. [1],[4]

Due to the presence of atypical lymphocytes in PLF, it may be mistaken for primary malignant cutaneous lymphomas, including small/medium-sized pleomorphic T-cell lymphoma and follicle center B-cell lymphoma, and pilotropic mycosis fungoides, etc., Marginal zone lymphoma is distinguished from PLF by diffuse proliferation of marginal zone cells, zones or sheets of plasma cells that are frequently monotypic, and the presence of reactive lymphoid follicles, while predilection for the face and solitariness help to distinguish PLF from pleomorphic small-/medium-sized T-cell lymphoma. The absence of atypical cells with phenotypically heterogeneous infiltrate in PLF allows its distinction from follicular mycosis fungoides. [2],[3],[5]

The lesions of PLF demonstrate benign behavior, though atypical lymphocytes can be seen in PLF. The lesions spontaneously disappear after incisional biopsy in a significant number of cases with solitory lesions. Various treatments like intra-lesional steroid injections, oral anti-malarials, methotrexate, cyclosporin, etc., have been reported with variable success rate. [2],[3],[4] They may be used in cases with residual or multiple lesions or large lesions like in the present case where excision may not be feasible.


   Conclusion Top


PLF is a distinctive cutaneous lymphoid hyperplasia. High-index of suspicion among dermatologist will avoid unnecessary use of oral antibiotics. Pathologist should differentiate PLF from primary malignant cutaneous lymphomas.

 
   References Top

1.Nakamura M, Kabashima K, Tokura Y. Pseudolymphomatous folliculitis presenting with multiple nodules. Eur J Dermatol 2009;19:263-4.  Back to cited text no. 1
    
2.Kakizaki A, Fujimura T, Numata I, Hashimoto A, Aiba S. Pseudolymphomatous folliculitis on the nose. Case Rep Dermatol 2012;4:27-30  Back to cited text no. 2
    
3.Lee HW, Ahn SJ, Lee MW, Choi JH, Moon KC, Koh JK. A case of pseudolymphomatous folliculitis. J Eur Acad Dermatol Venereol 2006;20:230-2  Back to cited text no. 3
    
4.Arai E, Okubo H, Tsuchida T, Kitamura K, Katayama I. Pseudolymphomatous folliculitis: A clinicopathologic study of 15 cases of cutaneous pseudolymphoma with follicular invasion. Am J Surg Pathol 1999;23:1313-9  Back to cited text no. 4
    
5.Dargent JL, Debois J, Sass U, Theunis A, André J, Simonart T. Unusual T cell pseudolymphoma with features of so-called pseudolymphomatous folliculitis. Dermatology 2002;204:159-61.  Back to cited text no. 5
    

What is new? 1. Pseudolymphomatous folliculitis is a distinctive type of cutaneous pseudolymphomas. 2. It may be under.reported due to low index of suspicion. 3. Identifying PLF will avoid unnecessary use of antibiotics.


    Figures

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

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[Pubmed] | [DOI]



 

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    Abstract
   Introduction
   Case Report
   Discussion
   Conclusion
    References
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