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What is cicatricial pemphigoid?

Cicatricial pemphigoid is an autoimmune disease that is characterised by blistering lesions on mucous membranes. It is also called benign mucous membrane pemphigoid or oral pemphigoid. Areas commonly involved are the oral mucosa (lining of the mouth) and conjunctiva (mucous membrane that coats the inner surface of the eyelids and the outer surface of the eye). Other areas that may be affected include the nostrils, oesophagus, trachea and genitals. Sometimes the skin may also be involved where blistering lesions can be found on the face, neck and scalp.

Brunsting Perry cicatricial pemphigoid is a rare variant in which localised crops of recurrent blisters arise within urticarial plaques, usually on the head and neck. The blisters may burst resulting in blood-crusted plaques and scars.

Who gets cicatricial pemphigoid?

Cicatricial pemphigoid is predominantly a disease of the elderly with a peak incidence at around 70 years. However, childhood cases have been reported. It appears to be twice as common in women than men.

What are the signs and symptoms of cicatricial pemphigoid?

Site Features
Eye
  • Sensation of grittiness or pain
  • Conjunctivitis
  • Lesions form, erode and heal to leave scar tissue
  • May lead to impaired vision or blindness
Mouth
  • Blisters form first on the gums near the teeth
  • Palate, tongue, lips, buccal mucosa, floor of the mouth and throat may be affected
  • Painful and make it difficult to eat
  • Lesions occurring in the throat (oesophagus, trachea and larynx) can become life-threatening
Skin
  • Blisters on the skin develop in 25-30% of patients
  • May be itchy
  • Bleeding may occur if traumatised
Nose
  • Nose bleeds after blowing the nose
  • Crusting causing discomfort
Genitals
  • Painful blisters and erosions on the clitoris, labia, shaft of the penis, perianal area

What causes cicatricial pemphigoid?

Cicatricial pemphigoid is an autoimmune blistering disease, which basically means that an individual’s immune systems starts reacting against his or her own tissue. In this particular instance autoantibodies react with proteins found in mucous membranes and skin tissue resulting in blistering lesions. The binding site appears to be within the anchoring filaments that help the epidermis (outside layer of skin) stick to the dermis (inner layer of skin).

Full article from DermNet NZ

http://www.dermnetnz.org/immune/cicatricial-pemphigoid.html

 

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In BP lesional skin, immunohistochemistry and confocal microscopy were performed for CD4+, CD25+, forkhead/winged helix transcription factor (FOXP3)+, transforming growth factor (TGF)-β+ and interleukin (IL)-10+ cells. In addition, the number of CD4+CD25++FOXP3+ Tregs in peripheral blood was assessed by flow cytometry, and the levels of TGF-β and IL-10 were determined in serum samples by enzyme-linked immunosorbent assay before and after steroid therapy. Controls included patients with psoriasis, atopic dermatitis (AD) and healthy donors.

The frequency of FOXP3+ cells was significantly reduced in skin lesions from patients with BP (P < 0.001) compared with psoriasis and AD. Moreover, the number of IL-10+ cells was lower in BP than in psoriasis (P < 0.001) and AD (P = 0.002), while no differences were observed in the number of TGF-β+ cells. CD4+CD25++FOXP3+

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Treg in the peripheral blood of patients with BP was significantly reduced compared with healthy controls (P < 0.001), and augmented significantly after steroid therapy (P = 0.001). Finally, TGF-β and IL-10 serum levels were similar in patients with BP compared with healthy controls. However, after therapy, BP patients showed significantly higher IL-10 serum levels than before therapy (P = 0.01).

Full article available at: http://onlinelibrary.wiley.com/doi/10.1111/jdv.12091/abstract;jsessionid=C37D521517222D9766F5D0D339765626.d04t01?deniedAccessCustomisedMessage=&userIsAuthenticated=false

Antiga, E., Quaglino, P., Volpi, W., Pierini, I., Del Bianco, E., Bianchi, B., Novelli, M., Savoia, P., Bernengo, M.G., Fabbri, P. and Caproni, M. (2013), Regulatory T cells in skin lesions and blood of patients with bullous pemphigoid. Journal of the European Academy of Dermatology and Venereology. doi: 10.1111/jdv.12091
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Associations between blood groups and several diseases are observed in the literature. Some of these have scientific support suggesting a rationale, statistical relation. The association between ABO groups with several malignancies, hypercholesterolemia, thrombosis, myocardial infarction, duodenal ulcer, infections, and autoimmune diseases is reported.  Pemphigus vulgaris (PV), a rare autoimmune, blistering disease, related to autoantibodies mainly directed to desmogleins, which lead to loss of keratinocyte adhesion.  The association between ABO groups and pemphigus has been proposed but not fully demonstrated. Shahkar et al., concluded the non-existent relationship between blood groups and the development of PV. The authors carried out a case-control study that showed there is no real association between blood groups and PV, in contrast to the work of Grob and Inderbitzin  and Altobella.  The authors determined that the presence of a particular blood group, in patients with the disease, does not vary significantly with “healthy” population distribution, which is very important because the relationship between blood groups and skin diseases has been controversial and not yet fully elucidated or explained clearly.

In 2007, Valikhani et al.,  showed not only that the ABO and Rhesus blood groups not have a particular distribution in the PV, other than the population, but no such relationship with any of the known variants of pemphigus, at least in Iran, suggesting the authors to conduct a study involving other areas of global demographic.

In Mexico, we conducted a similar study in a tertiary referral center for specialized dermatology consultation. We obtained the ABO and Rhesus blood groups of patients with PV in a period between January 2002 and October 2009, being our hospital a center that collects patients from different parts of Mexico and even South America.

We selected 70 charts of patients with PV. No differences in the presence of a particular blood group in patients with the disease were found (P=0.65). We sought to evaluate if any ABO group correlates with the clinical outcome (body surface area affected) of the patients studied. There were no positive or negative correlation between ABO groups and clinical outcome in PV (P=0.752)

We conclude, there is no association between ABO and Rhesus blood groups with PV, demonstrated by observing no differences between the presentations of a specific blood group in the disease. Moreover, there is no association between any ABO groups with the clinical outcome in PV.

Tirado-Sánchez A, Ponce-Olivera RM. Lack of relationship between blood groups and clinical outcome (body surface area affected) in patients with pemphigus vulgaris. Indian J Dermatol [serial online] 2012 [cited 2012 Sep 12];57:411-2. Available from: http://www.e-ijd.org/text.asp?2012/57/5/411/100513