Platelet-rich plasma (PRP).
Keywords : cutaneous pseudolymphoma. Primary cutaneous
lymphomas. Platelet-rich plasma (PRP).
Introduction:
Cutaneous pseudolymphoma (CPL), also called cutaneous lymphoid
hyperplasia, is a nonspecific disease that is characterized by a
polyclonal benign lymphoproliferative process1,2.
Pseudolymphoma can mimic lymphoma both clinically and histologically.1.2.3.4 Most cases are idiopathic. Known etiologies
include reaction to tattoo, arthropod bite, infection (borrelia
burgdorferi, molluscum contagiosum2, leishmaniasis),
vaccination and drugs (e.g., anticonvulsants).
Primary cutaneous lymphomas are a
heterogenous group of lymphoproliferative disorders that primarily
involve the skin. They are the most frequent extra nodal
lymphomas; classification is based on lymphocyte type and
includes B-cell and T-cell. Cutaneous T-cell lymphoma (CTCL) is the most
common type.6. Cutaneous B-cell lymphomas are
classified into four types: 1) primary cutaneous marginal zone B-cell
lymphoma (PCMZL) 2) primary cutaneous follicular center lymphoma (PCFCL)
3) primary cutaneous diffuse large B-cell lymphoma, leg type ( LBCL-L)
4) primary cutaneous large B-cell lymphoma,
other(LBCL-other)7,8
Platelet-rich
plasma (PRP) is a biological product defined as a portion of the plasma
fraction of autologous blood with a platelet concentration above the
baseline. It is obtained from the blood of patients collected before
centrifugation. Platelets contain growth factors and mediators in their
granules (TGF-B1, PDGF, bFGF, VEGF, EGF, IGF-1) and believed to promote
cell proliferation, differentiation, angiogenesis and chemotaxis. PRP is
considered a novel treatment modality especially for hair restoration
and skin rejuvenation, in combination with other modalities or
alone.9
Case presentation:
A 46-year-old man was referred to our hospital with complaint of
erythematous plaques on his head, especially the forehead, that started
20 years ago. Over time, the lesions had increased in size. the patient
had no remarkable past medical history or drug history. In 2009, a skin
biopsy was performed showing cutaneous B-cell pseudolymphoma (CBPL).
Immunohistochemistry (IHC) performed in the same year revealed CD20,
CD3, CD4, CD8, CD79a and BCL2 were positive and, no evidence of
lymphomatous involvement and no evidence of clonal staining for Kappa
and Lambda light chains. The patient was lost to follow up and treatment
until 2013, at which time repeat biopsy was performed and a diagnosis of
pseudolymphoma with dense lymphoid infiltration with germinal center
follicles was confirmed. After diagnosis, patient received 12 sessions
of monthly intralesional corticosteroids. Following treatment, the
patient’s lesions improved significantly and almost resolved. In 2017,
the patient had a hair transplant followed by a platelet-rich plasma
(PRP) session. One week after PRP, Following the first session of PRP,
lesions re-appeared on the head (FIGURE 1A). Re-biopsy from one of the
new lesions once again showed pseudolymphoma. The patient was re-treated
with intralesional corticosteroids and topical clobetasol, but was not
responsive to treatment. The patient also did not respond to treatment
with hydroxychloroquin and thalidomide.following enlargement of the
previous lesions, the biopsy was repeated, and atypical nodular and
diffuse lymphoid cell infiltration was seen, compatible with
lymphoproliferative disorder (FIGURE2A) and IHC study recommended. In
IHC, the finding (CD20 strongly positive and BCL2 and BCL6 positive) was
in favor of primary cutaneous follicular center lymphoma (PCFCL) (FIGURE
2 B,C,D,E). The patient did not have lymphadenopathy on examination and
paraclinical tests were normal. Also, chest, neck and abdominal and
pelvic CT scan were normal, consistent with being primary cutaneous
origin lymphoma. in the meantime, positive family history for Hodgkin
lymphoma in patient‘s sister revealed. Due to the lack of response to
previous treatment, the patient became candidate for rituximab (6-8
sessions every three weeks). At present, the patient has had a
significant improvement following receiving rituximab. (FIGURE 1 B)
Discussion:
In this study, a rare manner of cutaneous pseudolymphoma (CPL)with
non-ordinary progression time line that advanced into primary cutaneous
follicular center lymphoma is presented. This patient was confirmed case
of cutaneous B-cell pseudolymphoma (CBPL)for almost 4 years and had
complete response following intralesional corticosteroid therapy. But
just after hair transplantation and PRP sessions the disease recured,
and at this time following histopathology and immunohistochemistry the
primary follicular center B cell lymphoma was diagnosed. For us it was
an unanswered question that if these lesions represent actual
progression from cutaneous pseudo lymphoma to frank lymphoma as it has
been previously discussed in literature5 and also
Kulow et al reported a 4 patients initially diagnosed with combination
of clinical , histopathology and immunophenotyping as cutaneous
pseoudolymphoma and had a progressive clinical course evolving as
primary cutaneous lymphoma .4 or they were malignant
from beginning and not diagnosed properly which it seems unlikely due to
long duration of disease and complete response and clearance after
intralesional steroid therapy.
On the other hand, PCFCL is considered as the most common cutaneous
lymphoma12. While the clear pathogenesis of PCFCL has
remained unknown, Persistent antigenic stimulation has been suggested as
a cause for such a progression11. Thus, a long-time
follow-up is also absolutely necessary to make the final diagnosis.
In this study our case underwent various interventions including hair
transplantation and PRP. PRP
consist of growth factors and hormones which potentially could lead to
excess cell proliferation. It is unknown that these modalities alone or
their specific proliferative formulations may be the leading cause or
aggravating factor ending to primary cutaneous lymphoma specially for
patients with positive family history. According to recent reports12 and formation of malignancies after PRP ,it seems
wise to be cautious specially for patients with positive family or
personal history and further research should be done to investigate the
possibility and association of these factors.
•Acknowledgment:
This research has been supported by Tehran University of medical
sciences.
References:
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Figure legends:
Figure 1:
(A)Erythematous and indurated plaques and nodules on scalp, forehead
extending to vertex
(B) significant resolution of lesions after rituximab therapy.
Figure 2:
Primary cutaneous follicle center lymphoma, diffuse type (A-E).
(A)Diffuse and nodular growth pattern infiltrate of large lymphoid cells
with lack of mantle zone and tinigible body macrophages
(B)CD 10+ . (C) Bcl-6+. (D) CD 20+. (E) CD23+ meshwork pattern.