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Tham HL, Linder KE, Olivry T. Deep pemphigus (pemphigus vulgaris, pemphigus vegetans and paraneoplastic pemphigus) in dogs, cats and horses: a comprehensive review. BMC Vet Res 2020; 16:457. [PMID: 33228633 PMCID: PMC7686683 DOI: 10.1186/s12917-020-02677-w] [Citation(s) in RCA: 5] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 08/17/2020] [Accepted: 11/13/2020] [Indexed: 12/28/2022] Open
Abstract
Pemphigus is the term used to describe a group of rare mucocutaneous autoimmune bullous diseases characterized by flaccid blisters and erosions of the mucous membranes and/or skin. When the autoantibodies target desmosomes in the deep layers of the epidermis, deep pemphigus variants such as pemphigus vulgaris, pemphigus vegetans and paraneoplastic pemphigus develop. In this article, we will review the signalment, clinical signs, histopathology and treatment outcome of pemphigus vulgaris, pemphigus vegetans and paraneoplastic pemphigus in dogs, cats and horses; where pertinent, we compare the animal diseases to their human homologue. Canine, feline and equine pemphigus vulgaris, pemphigus vegetans and paraneoplastic pemphigus have many features similar to the human counterpart. These chronic and often relapsing autoimmune dermatoses require aggressive immunosuppressive therapy. In animals, the partial-to-complete remission of pemphigus vulgaris and pemphigus vegetans has been achieved with high dose glucocorticoid therapy, with or without adjunct immunosuppressants; the prognosis is grave for paraneoplastic pemphigus.
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Affiliation(s)
- Heng L. Tham
- Department of Small Animal Clinical Sciences, Virginia-Maryland College of Veterinary Medicine, Virginia Tech, Blacksburg, VA USA
| | - Keith E. Linder
- Comparative Medicine Institute, College of Veterinary Medicine, North Carolina State University, Raleigh, NC USA
- Department of Population Health and Pathobiology, College of Veterinary Medicine, North Carolina State University, Raleigh, NC USA
| | - Thierry Olivry
- Department of Clinical Sciences, College of Veterinary Medicine, North Carolina State University, Raleigh, NC USA
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2
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Shalin SC, Racher LM, Campbell KK. Lichenoid dermatoses involving the vulva: A clinical-pathologic correlation ✰. Semin Diagn Pathol 2020; 38:3-18. [PMID: 32951943 DOI: 10.1053/j.semdp.2020.09.005] [Citation(s) in RCA: 4] [Impact Index Per Article: 0.8] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 09/01/2020] [Accepted: 09/03/2020] [Indexed: 01/06/2023]
Abstract
The lichenoid tissue reaction pattern generally signifies cytotoxic damage to the epithelium. When such reaction pattern occurs on vulvar skin or mucosa, the effects can result in considerable morbidity. None of the entities discussed in this review are entirely unique to the vulva, however, some entities may classically occur at this site, while others tend to be widespread diseases that may incidentally affect vulvar skin and mucosa. Given the complex anatomy of the vulva and the bridging of a site showing both keratinizing squamous epithelium and non-keratinizing squamous mucosa, histopathologic features may display variation in presentation. Although identification of a "lichenoid reaction pattern" alone may provide insight into the disease process, understanding of clinical presentation and specific sites of involvement, along with recognition of the nuanced features of the disease entities can help establish a specific diagnosis. Accurate histopathologic diagnoses by pathologists can improve the ability for treating clinicians to implement timely and effective treatment.
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Affiliation(s)
- Sara C Shalin
- Department of Pathology, University of Arkansas for Medical Sciences, Little Rock, AR.
| | - Luann M Racher
- Department of Obstetrics and Gynecology, University of Arkansas for Medical Sciences, Little Rock, AR
| | - Katelynn K Campbell
- Department of Pathology, University of Arkansas for Medical Sciences, Little Rock, AR
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3
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Lepekhova AA, Teplyuk NP, Bolotova IM. Modern diagnostic methods of paraneoplastic pemphigus. VESTNIK DERMATOLOGII I VENEROLOGII 2019. [DOI: 10.25208/0042-4609-2019-95-5-7-16] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/02/2022] Open
Abstract
Paraneoplastic pemphigus (PNP) is one of the least investigated and rare forms of bullous dermatoses, which comes from underlying neoplasm. The article presents a literature review of dermatologist`s longstanding international experience about etiology, pathogenesis, diagnostics and treatment of this disease. According to the research results of native and foreign authors systematization of modern diagnostic methods of PNP and detectable antigens was performed.Conflict of interest: the authors state that there is no potential conflict of interest requiring disclosure in this article.
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Affiliation(s)
- A. A. Lepekhova
- I. M. Sechenov First Moscow State Medical University, Ministry of Health of the Russian Federation
| | - N. P. Teplyuk
- I. M. Sechenov First Moscow State Medical University, Ministry of Health of the Russian Federation
| | - I. M. Bolotova
- Pirogov Russian National Research Medical University, Ministry of Health of the Russian Federation
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4
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Asad U, Austin B, Sturgeon A, Stetson C. Paraneoplastic eczematous eruption associated with Hodgkin's lymphoma. Proc (Bayl Univ Med Cent) 2019; 32:587-588. [PMID: 31656430 DOI: 10.1080/08998280.2019.1644148] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 06/07/2019] [Revised: 07/07/2019] [Accepted: 07/12/2019] [Indexed: 01/12/2023] Open
Abstract
Hodgkin's disease (HD) is a common malignant lymphoma that classically presents with painless lymphadenopathy, weight loss, nocturnal sweats, fever, and generalized pruritus. Unlike other lymphoproliferative disorders, cutaneous involvement is uncommon with HD. Diffuse hyperpigmentation, prurigo nodularis, acquired ichthyosis, erythema nodosum, mycosis fungoides, herpes zoster, and vasculitis have all been described as nonspecific or paraneoplastic manifestations of HD. A small set of cases of adult new-onset recalcitrant eczema has recently been described as a nonspecific cutaneous manifestation of HD. In agreement with these reports, we present a case of a 28-year-old woman who initially presented with persistent lymphadenopathy and a pruritic eczematous dermatitis that preceded the diagnosis of HD.
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Affiliation(s)
- Usman Asad
- School of Medicine, Texas Tech University Health Sciences CenterLubbockTexas
| | - Brett Austin
- Department of Dermatology, Texas Tech University Health Sciences CenterLubbockTexas
| | - Ashley Sturgeon
- Department of Dermatology, Texas Tech University Health Sciences CenterLubbockTexas
| | - Cloyce Stetson
- Department of Dermatology, Texas Tech University Health Sciences CenterLubbockTexas
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5
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DiSantis F, Hames N, Weiss L. A Teenager With Painful Oral and Genital Lesions. Clin Pediatr (Phila) 2019; 58:1034-1037. [PMID: 31113215 DOI: 10.1177/0009922819850478] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 11/16/2022]
Affiliation(s)
- Francesca DiSantis
- 1 Philadelphia College of Osteopathic Medicine Georgia Campus, Suwanee, GA, USA
| | - Nicole Hames
- 2 Department of Pediatrics, Emory University School of Medicine, Atlanta, GA, USA.,3 Children's Healthcare of Atlanta, Atlanta, GA, USA
| | - Lindsay Weiss
- 2 Department of Pediatrics, Emory University School of Medicine, Atlanta, GA, USA.,3 Children's Healthcare of Atlanta, Atlanta, GA, USA
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6
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Jain A, Prakash G, Nampoothiri RV, De D, Bal A, Khadwal A, Lad D, Malhotra P, Varma S. Peri-anal Paraneoplastic Pemphigus Heralding the Relapse of Follicular Lymphoma and Its Successful Management by Rituximab: A Short Correspondence. Indian J Hematol Blood Transfus 2016; 32:519-521. [PMID: 27812271 DOI: 10.1007/s12288-016-0689-4] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 04/16/2016] [Accepted: 05/30/2016] [Indexed: 11/26/2022] Open
Affiliation(s)
- Ankur Jain
- Department of Hematology, PGIMER Chandigarh, Chandigarh, India
| | - Gaurav Prakash
- Department of Hematology, PGIMER Chandigarh, Chandigarh, India
| | | | - Dipankar De
- Department of Dermatology, PGIMER Chandigarh, Chandigarh, India
| | - Amanjit Bal
- Department of Histopathology, PGIMER Chandigarh, Chandigarh, India
| | - Alka Khadwal
- Department of Hematology, PGIMER Chandigarh, Chandigarh, India
| | - Deepesh Lad
- Department of Hematology, PGIMER Chandigarh, Chandigarh, India
| | - Pankaj Malhotra
- Department of Hematology, PGIMER Chandigarh, Chandigarh, India
| | - Subhash Varma
- Department of Hematology, PGIMER Chandigarh, Chandigarh, India
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7
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Huang LC, Wong JR, Alonso-Llamazares J, Nousari CH, Perez VL, Amescua G, Karp CL, Galor A. Pseudopemphigoid as caused by topical drugs and pemphigus disease. World J Ophthalmol 2015; 5:1-15. [DOI: 10.5318/wjo.v5.i1.1] [Citation(s) in RCA: 5] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 05/22/2014] [Revised: 09/19/2014] [Accepted: 11/10/2014] [Indexed: 02/05/2023] Open
Abstract
Pseudopemphigoid can cause a chronic cicatricial conjunctivitis that is clinically identical to the manifestations seen in mucous membrane pemphigoid, a disorder with a common clinical phenotype and multiple autoimmune links. For the purpose of this review, we will describe pseudopemphigoid as caused by topical drugs, the most common etiology with ocular manifestations, and as caused by the pemphigus disease, a more rare etiology. Specifically, we will discuss the ophthalmological features of drug-induced cicatricial conjunctivitis, pemphigus vulgaris, and paraneoplastic pemphigus. Other etiologies of pseudopemphigoid exist that will not be described in this review including autoimmune or inflammatory conditions such as lichen planus, sarcoidosis, granulomatosis with polyangiitis (Wegener’s granulomatosis), erythema multiforme (minor, major, and Stevens-Johnson syndrome), bullous pemphigoid, skin-dominated linear IgA bullous dermatosis, and skin-dominated epidermolysis bullosa acquisita. Prompt diagnosis of the underlying etiology in pseudopemphigoid is paramount to the patient’s outcome as certain diseases are associated with a more severe clinical course, increased ocular involvement, and differential response to treatment. A complete history and ocular examination may find early cicatricial changes in the conjunctiva that are important to note and evaluate to avoid progression to more severe disease manifestations. When such cicatricial changes are noted, proper diagnostic techniques are needed to help elucidate a diagnosis. Lastly, collaboration between ophthalmologists and subspecialists such as dermatologists, pathologists, immunologists, and others involved in the care of the patient is needed to ensure optimal management of disease.
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8
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Al Zamel G, Micheletti RG, Nasta SD, Palakshappa J, Stoopler ET. The importance of multidisciplinary healthcare for paraneoplastic pemphigus. SPECIAL CARE IN DENTISTRY 2014; 35:143-7. [PMID: 25257550 DOI: 10.1111/scd.12093] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/24/2022]
Abstract
Paraneoplastic pemphigus (PNP) is a mucocutaneous disease that occurs in association with an underlying neoplasm. Oral mucosal lesions may be the only manifestation of this condition, or they may be observed in association with cutaneous lesions. The prognosis of PNP is generally poor, and the disease is often fatal. This article highlights an aggressive case of PNP that initially presented with oral mucosal lesions and emphasizes the importance of a multidisciplinary approach for evaluation and management of this condition.
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Affiliation(s)
- Ghada Al Zamel
- Former Resident, Department of Oral Medicine, University of Pennsylvania School of Dental Medicine, Philadelphia, Pennsylvania
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Lehman VT, Barrick BJ, Pittelkow MR, Peller PJ, Camilleri MJ, Lehman JS. Diagnostic imaging in paraneoplastic autoimmune multiorgan syndrome: retrospective single site study and literature review of 225 patients. Int J Dermatol 2014; 54:424-37. [DOI: 10.1111/ijd.12603] [Citation(s) in RCA: 14] [Impact Index Per Article: 1.3] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 12/12/2022]
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10
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Otten JV, Hashimoto T, Hertl M, Payne AS, Sitaru C. Molecular diagnosis in autoimmune skin blistering conditions. Curr Mol Med 2014; 14:69-95. [PMID: 24160488 PMCID: PMC3905716 DOI: 10.2174/15665240113136660079] [Citation(s) in RCA: 70] [Impact Index Per Article: 6.4] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 01/07/2013] [Revised: 03/12/2013] [Accepted: 06/04/2013] [Indexed: 12/22/2022]
Abstract
Blister formation in skin and mucous membranes results from a loss of cell-cell or cell-matrix adhesion and is a common outcome of pathological events in a variety of conditions, including autoimmune and genetic diseases, viral and bacterial infections, or injury by physical and chemical factors. Autoantibodies against structural components maintaining cell-cell and cell-matrix adhesion induce tissue damage in autoimmune blistering diseases. Detection of these autoantibodies either tissue-bound or circulating in serum is essential to diagnose the autoimmune nature of disease. Various immunofluorescence methods as well as molecular immunoassays, including enzyme-linked immunosorbent assay and immunoblotting, belong to the modern diagnostic algorithms for these disorders. There is still a considerable need to increase awareness of the rare autoimmune blistering diseases, which often show a severe, chronic-relapsing course, among physicians and the public. This review article describes the immunopathological features of autoimmune bullous diseases and the molecular immunoassays currently available for their diagnosis and monitoring.
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Affiliation(s)
| | | | | | | | - C Sitaru
- Department of Dermatology, University of Freiburg, Hauptstrasse 7, D-79104 Freiburg, Germany.
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11
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Gissi DB, Bernardi A, D'Andrea M, Montebugnoli L. Paraneoplastic pemphigus presenting with a single oral lesion. BMJ Case Rep 2013. [PMID: 23283620 DOI: 10.1136/bcr-2012-007771.] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Abstract] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/04/2022] Open
Abstract
Paraneoplastic pemphigus (PNP) is recognised in most cases after diagnosis of malignant and benign haematological tumours. PNP usually presents with severe and diffuse oral ulcerations, ocular lesions, lichen planus-like skin lesions and frequently genital ulcerations. We describe the uncommon case of a patient unaware of any neoplasia with a unique ulcerated oral lesion with histological (acantholysis of the basal epithelial layer, necrotic keratinocytes and pronounced regenerative hyperplasia) and immunofluorescent (direct immunofluorescence test exhibited immunoglobulin IgG, fibrinogen and C3 deposition in intercellular areas and along the basement membrane; indirect immunofluorescence test performed on rat bladder showed bright fluorescence) features suggestive of PNP. Diagnosis of PNP was strengthened by the subsequent discovery of monoclonal gammopathy. The reported case is quite unusual if we consider the clinical appearance of the oral lesions and the patient's negative medical history. Following serological examinations, the patient proved to have monoclonal gammopathy of undetermined significance (MGUS), one of the most common premalignant plasma cell disorders.
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Affiliation(s)
- Davide Bartolomeo Gissi
- Section of Oral Science, Department of Biomedical and Neuro-Muscular Sciences, University of Bologna, Bologna, Italy
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12
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Gissi DB, Bernardi A, D'Andrea M, Montebugnoli L. Paraneoplastic pemphigus presenting with a single oral lesion. BMJ Case Rep 2013; 2013:bcr-2012-007771. [PMID: 23283620 DOI: 10.1136/bcr-2012-007771] [Citation(s) in RCA: 6] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/03/2022] Open
Abstract
Paraneoplastic pemphigus (PNP) is recognised in most cases after diagnosis of malignant and benign haematological tumours. PNP usually presents with severe and diffuse oral ulcerations, ocular lesions, lichen planus-like skin lesions and frequently genital ulcerations. We describe the uncommon case of a patient unaware of any neoplasia with a unique ulcerated oral lesion with histological (acantholysis of the basal epithelial layer, necrotic keratinocytes and pronounced regenerative hyperplasia) and immunofluorescent (direct immunofluorescence test exhibited immunoglobulin IgG, fibrinogen and C3 deposition in intercellular areas and along the basement membrane; indirect immunofluorescence test performed on rat bladder showed bright fluorescence) features suggestive of PNP. Diagnosis of PNP was strengthened by the subsequent discovery of monoclonal gammopathy. The reported case is quite unusual if we consider the clinical appearance of the oral lesions and the patient's negative medical history. Following serological examinations, the patient proved to have monoclonal gammopathy of undetermined significance (MGUS), one of the most common premalignant plasma cell disorders.
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Affiliation(s)
- Davide Bartolomeo Gissi
- Section of Oral Science, Department of Biomedical and Neuro-Muscular Sciences, University of Bologna, Bologna, Italy
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13
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Abstract
Pemphigus is an autoimmune bullous disease, in which autoantibodies react with the cell–cell adhesion structures, desmosomes, causing blisters and erosions on the oral mucosa and skin. Pemphigus is divided into two major subtypes: pemphigus vulgaris and pemphigus foliaceus. Oral corticosteroids are the primary treatment modality for pemphigus, while other therapeutic options, such as steroid pulse therapy, immunosuppressants, intravenous immunoglobulins, plasmapheresis and anti-CD20 monoclonal antibody therapy, are occasionally employed. Immunosuppressants used to treat pemphigus include azathioprine, cyclophosphamide, cyclosporine, mycophenolate mofetil and mizoribine. In this review, we summarize the current concepts of immunotherapy for the treatment of pemphigus in the Japanese population.
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Affiliation(s)
- Daisuke Tsuruta
- Department of Dermatology, Kurume University School of Medicine, and Kurume University Institute of Cutaneous Cell Biology, 67 Asahimachi, Kurume, Fukuoka 830-0011, Japan
- Department of Dermatology, Osaka City University Graduate School of Medicine, Osaka, Japan
| | - Norito Ishii
- Department of Dermatology, Kurume University School of Medicine, and Kurume University Institute of Cutaneous Cell Biology, 67 Asahimachi, Kurume, Fukuoka 830-0011, Japan
| | - Takashi Hashimoto
- >Department of Dermatology, Kurume University School of Medicine, and Kurume University Institute of Cutaneous Cell Biology, 67 Asahimachi, Kurume, Fukuoka 830-0011, Japan
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14
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Abstract
The presence of one autoimmune disorder helps lead to the discovery of other autoimmune conditions. It is thought that diseases in which autoimmunity is a feature tend to be associated together more often than one can ascribe to chance. A variety of diseases have been implicated in the onset of intraepidermal and subepidermal autoimmune diseases. The presence of one autoimmune disease should alert the physician to watch for a second immunologic disorder. A list of autoimmune bullous diseases associations includes autoimmune bullous diseases, pemphigus, pemphigoid, epidermolysis bullosa acquisita, dermatitis herpetiformis (Duhring), linear immunoglobulin A disease, and multiple autoimmune syndrome.
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Affiliation(s)
- Suzana Ljubojevic
- University Department of Dermatology and Venereology, University Hospital Center Zagreb, School of Medicine,University of Zagreb, Croatia.
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15
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Abstract
Vesicobullous and inflammatory diseases produce vulvar erosions that may exhibit nonspecific morphology and represent a diagnostic challenge. An approach to arriving at the correct diagnosis is presented. Most common etiologies are reviewed.
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Affiliation(s)
- Clare Pipkin
- Department of Dermatology, Duke University, Durham, NC 27710, USA.
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16
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Tam PMK, Cheng LL, Young AL, Lam PTH. Paraneoplastic pemphigus: an uncommon cause of chronic cicatrising conjunctivitis. BMJ Case Rep 2009; 2009:bcr12.2008.1306. [PMID: 21998619 DOI: 10.1136/bcr.12.2008.1306] [Citation(s) in RCA: 14] [Impact Index Per Article: 0.9] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 01/27/2023] Open
Abstract
We report a case of paraneoplastic pemphigus (PNP) as an uncommon but severe cause of cicatrising conjunctivitis. Initially diagnosed as drug eruptions, the patient's condition did not improve despite cessation of chemotherapy. Immunohistological confirmation of PNP has led to the use of combined oral prednisolone and intravenous immunoglobulin. Her ocular conditions stabilised with complete recovery of vision. PNP is a rare disease that can present with ocular involvement. Ophthalmologists should play an active role in monitoring and treatment of ocular surface complications such as symblepharon formation, severe dry eye and epithelial breakdown. Vigorous and prompt treatment is the key to successful prevention of irreversible and blinding complications. The atypical feature in this case is the presence of eosinophilic infiltration on histology that is a feature of allergic aetiologies rather than classical PNP.
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Affiliation(s)
- Patrick Mang Kwan Tam
- Prince of Wales Hospital, Ophthalmology and Visual Sciences, 7th Floor, Block B Staff Quarters, Shatin, Hong Kong
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de Souza Azevedo R, Abrahão AC, de Albuquerque EB. Synchronous orofacial granulomatosis and mucoepidermoid carcinoma: paraneoplastic syndrome or coincidence? ACTA ACUST UNITED AC 2008; 106:e40-5. [PMID: 18554941 DOI: 10.1016/j.tripleo.2008.04.014] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 01/08/2008] [Revised: 04/13/2008] [Accepted: 04/17/2008] [Indexed: 01/10/2023]
Abstract
Some malignant neoplasias induce the appearance of local or systemic manifestations at distant sites, which can act as indicators of their presence in a process named paraneoplastic syndrome. Granulomatous reactions have already been described related to malignancies. This report describes a case of synchronous orofacial granulomatosis and mucoepidermoid carcinoma and discusses the significance of this association.
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Affiliation(s)
- Rebeca de Souza Azevedo
- Lato Sensu Postgraduate Program, Stomatology Specialization, Department of Oral Pathology and Diagnosis, School of Dentistry, Federal University of Rio de Janeiro, Rio de Janeiro, Brazil
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Lo Russo L, Fedele S, Guiglia R, Ciavarella D, Lo Muzio L, Gallo P, Di Liberto C, Campisi G. Diagnostic pathways and clinical significance of desquamative gingivitis. J Periodontol 2008; 79:4-24. [PMID: 18166088 DOI: 10.1902/jop.2008.070231] [Citation(s) in RCA: 73] [Impact Index Per Article: 4.3] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/13/2022]
Abstract
The term desquamative gingivitis (DG) refers to a clinical manifestation that can be caused by several disorders. Many of them are immunologically mediated; in addition to the oral cavity, they can affect extraoral mucocutaneous sites, e.g., larynx, conjunctiva, esophagus, nasal and genital mucosa, and the skin. The degree of oral, periodontal, and systemic involvement determines the overall morbidity and, sometimes, the mortality of these disorders. We comprehensively review disorders commonly associated with DG and highlight diagnostic pathways, guidelines for differential diagnosis, and oral, periodontal, and systemic implications. More rare conditions are reviewed as well. Mucous membrane pemphigoid, oral lichen planus, and pemphigus vulgaris are responsible for the majority of cases of DG. In addition, other uncommon disorders should be considered. Accurate clinical, histologic, and serologic investigations are often required to differentiate among DG-associated disorders, provide adequate therapy, and improve the prognosis of patients.
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Affiliation(s)
- Lucio Lo Russo
- Department of Oral Sciences, Faculty of Medicine, School of Dentistry, Oral Medicine Section, University of Palermo, Palermo, Italy.
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19
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Woo VL, Abdelsayed R. Oral manifestations of internal malignancy and paraneoplastic syndromes. Dent Clin North Am 2008; 52:203-x. [PMID: 18154871 DOI: 10.1016/j.cden.2007.09.005] [Citation(s) in RCA: 9] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 05/25/2023]
Abstract
Malignant tumors of visceral organs are a fundamental feature of familial cancer and paraneoplastic syndromes. In many instances, the presence of an internal and often occult malignancy may be forewarned by various external manifestations. Several of these findings are preferentially localized to the head and neck region, including the oral cavity proper. This places the dental practitioner in a unique position to detect these "markers" of occult neoplastic involvement. Because these markers may present before an established syndrome or cancer diagnosis, even representing the first expression of disease in some cases, early recognition by a dentist may lead to timely diagnosis and management of these cancer-associated syndromes.
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Affiliation(s)
- Victoria L Woo
- Columbia University College of Dental Medicine, 630 West 168th Street, PH 1562 West, New York, NY 10032, USA.
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20
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Abstract
Paraneoplastic pemphigus (PNP) is a life-threatening autoimmune blistering skin disease. Clinically, it is characterized by severe mucosal erosions and various cutaneous lesions associated with lymphoproliferative neoplasmas. Suprabasal acantholysis and clefts with scattered necrotic keratinocytes are the unique histopathological features. PNP patient sera recognize multiple antigens, which have been identified as the plakin protein family that includes desmoplakin, bullous pemphigoid antigen I (BPAG1), envoplakin and periplakin, and desmogleins 1 and 3. Castleman's tumor, non-Hodgkin's lymphoma, thymoma, follicular dendritic cell sarcoma and chronic lymphocytic leukemia are the commonly associated neoplasmas in PNP. We have also demonstrated that the autoantibodies reacting to epidermal proteins are directly produced by the cells in the associated tumors. Bronchiolitis obliterans is frequently found in PNP and may cause respiratory failure and death. In our experience, the early detection and removal of the tumor and i.v. administration of immunoglobulin are critical for the treatment of PNP.
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Affiliation(s)
- Xuejun Zhu
- Department of Dermatology, Peking University First Hospital, Beijing, China.
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Sirvain S, Crépeau T, Barré E, Garrido JF, Hallé O. Une gingivostomatite traînante. Rev Med Interne 2006; 27:789-90. [PMID: 16790298 DOI: 10.1016/j.revmed.2006.04.008] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 03/24/2006] [Accepted: 04/06/2006] [Indexed: 11/28/2022]
Affiliation(s)
- S Sirvain
- Service de médecine interne 2, CHG d'Alès, BP 139, 811, avenue Jean-Goubert, 30103 Alès cedex, France.
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