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Yohannis Derbew Molla, Isak Omer Answar, Biruk Mulat Worku, Amanuel Kassa Tadesse, Elias Manaye Tefera, Bewketu Abebe Alemu, Gebrehiwot Lema Legese, Samuel Addisu Abera
INTRODUCTION AND IMPORTANCE: Paraneoplastic pemphigus (PNP) is an uncommon autoimmune mucocutaneous disease characterized by severe stomatitis, polymorphous skin eruptions, and the presence of underlying neoplasms. Unique histopathological features include suprabasal acantholysis and clefts with scattered necrotic keratinocytes. CASE PRESENTATION: A 27-year-old female patient presented with a 3-month history of a painless swelling, approximately the size of a pea, on the left lateral aspect of her neck and axillary area...
April 2024: Annals of Medicine and Surgery
#2
Jacqueline A Turner, Ali Hakimi, Hannah Lee, Jeffrey T Schowinsky, Jeffrey M Sippel, Bradford J Siegele, Raul M Torres, William A Robinson
The Castleman triad has been described in a select few patients presenting with a retroperitoneal mass, mucocutaneous pemphigus vulgaris, and bronchiolitis obliterans. Here, we describe the Castleman triad in a 19-year-old male with unicentric hyaline vascular type Castleman disease (HV-CD). This patient presented with an array of positive antibodies, including anti-cyclic citrullinated peptide, anti-double-stranded DNA, and Sjogren's IgG. Interestingly, the patient's rheumatologic symptoms resolved after tumor resection, while his antibody profile remained relatively unchanged...
2024: Case Reports in Immunology
#3
Elena Pastukhova, Feras M Ghazawi
COVID-19 has been implicated in various cutaneous autoimmune diseases. Pemphigus is a group of autoimmune blistering diseases that target the desmosomal complexes. Pemphigus triggered by COVID-19 has been seldom reported in the literature and remains both a diagnostic and therapeutic challenge. We report a case of COVID-19-induced pemphigus that responded well to prednisone and mycophenolate mofetil after 9 months from initial presentation. On histologic examination, both intercellular and basement membrane staining were noted...
2024: SAGE Open Medical Case Reports
#4
REVIEW
Yefei Zhang, Jiahui Chen, Jia Tian, Yehui Zhou, Yan Liu
Plakophilin 3 (PKP3), a component of desmosome, is aberrantly expressed in many kinds of human diseases, especially in cancers. Through direct interaction, PKP3 binds with a series of desmosomal proteins, such as desmoglein, desmocollin, plakoglobin, and desmoplakin, to initiate desmosome aggregation, then promotes its stability. As PKP3 is mostly expressed in the skin, loss of PKP3 promotes the development of several skin diseases, such as paraneoplastic pemphigus, pemphigus vulgaris, and hypertrophic scar...
December 4, 2023: Cancer Science
#5
JOURNAL ARTICLE
Xin Ma, Jiyuan Li, Linlin Fan, Hongwei Jiang, Gaishao Shi, Dongfeng Ge, Xiaofei Shi
BACKGROUND: The literature describes a case of systemic lupus erythematosus (SLE) complicated with Castleman's disease (CD) and secondary paraneoplastic pemphigus (PNP). CASE PRESENTATION: A 12-year-old female presented with a neck mass, rash, arthralgia, and skin and mouth ulceration for 5 years were admitted. All blood cells were low. Multiple autoantibodies associated with SLE were positive. The pathology of the neck mass revealed the classical manifestations of CD...
October 19, 2023: Pediatric Rheumatology Online Journal
#6
JOURNAL ARTICLE
Nishita Lockwood, Shaman Bhullar, Gauri Panse, Jonathan S Leventhal
No abstract text is available yet for this article.
November 2023: JAAD Case Reports
#7
JOURNAL ARTICLE
Sai Wang, Rui Wang, Panpan Shang, Xuejun Zhu, Xixue Chen, Guohong Zhang, Mingyue Wang
Unicentric Castleman disease (UCD) is a rare lymphoproliferative disorder. Paraneoplastic pemphigus (PNP) is a major complication associated with poor UCD prognosis. However, the genomic profiles and prognostic biomarkers of PNP-associated UCD remain unclear. Here, we performed whole-exome sequencing analysis for 28 matched tumor-normal pairs, and nine tumor-only samples to define the genomic landscape of Chinese patients with PNP-associated UCD. An integrative analysis was performed to identify somatic mutations, the mutational signatures, and key pathways in tumors...
October 13, 2023: Journal of Investigative Dermatology
#8
JOURNAL ARTICLE
Latifah Albrahim, Amal Aboud Alasmari, Majed Aleissa
Pemphigus vulgaris (PV) is an autoimmune blistering disease affecting the skin and mucosa. It clinically presents as painful erosions, mainly in the oral cavity, and flaccid blisters and erosions on the skin. Steven-Johnson syndrome/toxic epidermal necrolysis (SJS/TEN) is a severe mucocutaneous drug hypersensitivity reaction characterized by painful, dusky, erythematous eruptions that often progress to blisters, erosions, and extensive epidermal detachment accompanied by systemic symptoms. Herein, we report the case of a 48-year-old man who presented with extensive skin and mucosal lesions following the ingestion of an unknown medication...
September 12, 2023: Dermatology Reports
#9
JOURNAL ARTICLE
Meiyu Guo, Lin Nong, Mingyue Wang, Yang Zhang, Lihong Wang, Yuhua Sun, Qingyun Wang, Huihui Liu, Jinping Ou, Xinan Cen, Hanyun Ren, Yujun Dong
The purpose is to ascertain the clinical impact of Castleman disease (CD) by reassessment of the real-world data from Peking University First Hospital (PKUFH). The results will contribute to the standardization of diagnosis and treatment on CDs. Based on the last 15-year retrospective real-world data from Peking University First Hospital (PKUFH), we reclassified and re-evaluated the clinical and pathological information of patients with pathologically suspected diagnosis of CD. A total of 203 patients were included in our study, in which the diagnosis of CD was confirmed in 189 cases, including 118 patients with unicentric CD (UCD, n = 118, 62...
September 26, 2023: Annals of Hematology
#10
Hajar Moata, Fouzia Hali, Safia Zafad, Hassan Jouhadi, Soumiya Chiheb
Paraneoplastic pemphigus (PNP) is a rare, autoimmune, blistering condition defined by severe stomatitis, polymorphous cutaneous eruptions, and underlying neoplasms. PNP associated with solid cancer is extremely rare. An association with prostate adenocarcinoma remains exceptional. We describe a 69-year-old patient with recalcitrant PNP associated with prostate adenocarcinoma showing spectacular response immediately after associating hormonotherapy with conventional immunosuppressive drugs.
August 2023: Curēus
#11
REVIEW
Simo Huang, Hannah J Anderson, Jason B Lee
In the second part of this Continuing Medical Education article on paraneoplastic pemphigus/paraneoplastic autoimmune multiorgan syndrome (PNP/PAMS), its diagnostic criteria, investigative work-up, and management are reviewed. PNP/PAMS is a rare autoimmune blistering disorder associated with high morbidity and mortality. Recognizing PNP/PAMS's key features and its diagnostic criteria is critical in initiating appropriate work-up. Evaluating PNP/PAMS requires knowledge of its findings on histopathology, direct immunofluorescence, indirect immunofluorescence, and enzyme-linked immunosorbent assay...
September 13, 2023: Journal of the American Academy of Dermatology
#12
Emiliano Antiga, Rikke Bech, Roberto Maglie, Giovanni Genovese, Luca Borradori, Barbara Bockle, Marzia Caproni, Frédéric Caux, Nisha Suyien Chandran, Alberto Corrà, Francesco D'Amore, Maryam Daneshpazhooh, Dipankar De, Dario Didona, Marian Dmochowski, Kossara Drenovska, Jan Ehrchen, Claudio Feliciani, Matthias Goebeler, Richard Groves, Claudia Günther, Sanjeev Handa, Silke C Hofmann, Barbara Horvath, Dimitrios Ioannidis, Hana Jedlickova, Cezary Kowalewski, Khalaf Kridin, Pascal Joly, Yen Loo Lim, Branka Marinovic, Emanual Maverakis, Joost Meijer, Aikaterini Patsatsi, Carlo Pincelli, Catherine Prost, Jane Setterfield, Eli Sprecher, Dusan Skiljevic, Kaisa Tasanen, Soner Uzun, Nina Van Beek, Snejina Vassileva, Artem Vorobyev, Igor Vujic, Gang Wang, Mingyue Wang, Katarzyna Wozniak, Savas Yayli, Giovanna Zambruno, Takashi Hashimoto, Enno Schmidt, José Manuel Mascarò, Angelo Valerio Marzano
No abstract text is available yet for this article.
November 2023: Journal of the European Academy of Dermatology and Venereology: JEADV
#13
JOURNAL ARTICLE
Vivek Kumar Garg, Harpal S Buttar, Sajad Ahmad Bhat, Nuftieva Ainur, Tannu Priya, Dharambir Kashyap, Hardeep Singh Tuli
Both Stevens-Johnson syndrome (SJS) and Toxic-epidermal necrolysis (TEN) are generally medication-induced pathological conditions that mostly affect the epidermis and mucus membranes. Nearly 1 to 2 patients per 1,000,000 population are affected annually with SJS and TEN, and sometimes these maladies can cause serious life-threatening events. The reported death rates for SJS range from 1 to 5%, and 25 to 35% for TEN. The mortality risk may even be higher among elderly patients, especially in those who are affected by a significant amount of epidermal detachment...
August 21, 2023: Recent advances in inflammation & allergy drug discovery
#14
REVIEW
Hannah J Anderson, Simo Huang, Jason B Lee
Paraneoplastic pemphigus/paraneoplastic autoimmune multiorgan syndrome (PNP/PAMS) is a highly fatal autoimmune blistering disease. The condition occurs in patients with underlying benign or malignant neoplasms, most commonly lymphoproliferative disorders. Both humoral and cell-mediated immunities contribute to the pathogenesis, and autoantibodies against plakin family proteins are characteristic. Patients typically present with severe stomatitis and polymorphous skin lesions, which are often resistant to treatment...
August 17, 2023: Journal of the American Academy of Dermatology
#15
JOURNAL ARTICLE
Marta Kurzeja, Malgorzata Olszewska, Andrzej Grzybowski, Lidia Rudnicka
Autoimmune bullous diseases represent a heterogenous group of disorders caused by autoantibodies against adhesion molecules; the location of the target protein determines the level of cleft formation. The spectrum of ocular lesions in autoimmune bullous diseases can range from mild symptoms to severe involvement with sight impairment and even, in some cases, blindness. In pemphigus vulgaris, the prevalence of ocular involvement has been reported to be between 7% and 26%. The most common clinical sign of ocular pemphigus vulgaris is bilateral conjunctivitis with hyperemia...
August 14, 2023: Clinics in Dermatology
#16
Jimena Laiseca-García, Carmen García Rodríguez, Estefanía Moreno-Mata
We report the case of a woman who started with a lichenoid eruption, unfavorable evolution, for which a drug reaction was suspected. The final diagnosis was paraneoplastic pemphigus. Multidisciplinary care and evaluation by an Allergist is important in patients with severe skin reactions, suspected of drug reactions, due to the difficulty in establishing the diagnosis.
June 28, 2023: Revista Alergia Mexico: Organo Oficial de la Sociedad Mexicana de Alergia e Inmunología, A.C
#17
Kanako Hashimoto, Kenta Kobayashi, Yoshio Nakamura, Yuriko Yukino, Gonogi Tsuji, Haryoon Kim, Keisuke Kataoka, Masayuki Amagai
No abstract text is available yet for this article.
August 2023: Australasian Journal of Dermatology
#18
EDITORIAL
Alvise Sernicola, Mauro Alaibac
No abstract text is available yet for this article.
2023: Frontiers in Medicine
#19
Jonathan S Emerson, Mark Schifter, Pei Dai, Jocelyn Jiang, Mark S Taylor, Michelle Kang, Kenelm Kwong, Hadleigh Clark, Suzanne Cullican, David Campbell, Ming-Wei Lin
We present a series of five cases who presented to our institution with treatment-refractory mucosal ulceration, all of whom were subsequently diagnosed with paraneoplastic pemphigus (PNP). This case series highlights the diagnostic and treatment considerations for PNP - in particular, the steroid-dependent, recalcitrant, polymorphic manifestations; the combination of histopathological and clinical findings that may overlap with clinically similar diseases, for example, pemphigus vulgaris and lichen planus; the importance of immunopathological findings for its diagnosis, and the need for surveillance and management of life-threatening bronchiolitis obliterans...
June 2023: Skin Health Dis
#20
JOURNAL ARTICLE
Yannick Dieudonné, Marc-Antoine Silvestrini, Antoine Dossier, Véronique Meignin, Fanélie Jouenne, Thibault Mahévas, Jean-David Bouaziz, Margaret A Jackson, Pierre Mordant, Justine Poirot, Fanny Onodi, Julien Calvani, Muriel Hourseau, Diane Evrard, Mirlinda Berisha, François Perrin, Claire Danel, Raphael Borie, Lionel Galicier, Samia Mourah, Djaouida Bengoufa, Eric Oksenhendler, Sabine Grootenboer-Mignot, David Boutboul
Unicentric Castleman disease (UCD) is a lymphoproliferative disease of unknown cause. Paraneoplastic pemphigus (PNP) is a major complication shown to be associated with a poor prognosis, with particular severity in patients with bronchiolitis obliterans (BO). This study describes the clinical and biological characteristics of UCD-PNP patients in a large Western cohort. A total of 148 patients diagnosed with UCD were identified, including 14 patients with a defined PNP. PNP was significantly associated with myasthenia gravis (MG) and FDC sarcoma during follow-up (FDCS)...
July 2023: British Journal of Haematology
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