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Case Report
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| Refractory bilateral scrotal ulcers with numerous IgG4-positive plasma cells: Another skin location for IgG4-related sclerosing disease | ||||||
| Jaclyn L Jerz1, Jae Y Ro2, Alberto G Ayala3 | ||||||
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1MD, Pathology Resident; Department of Pathology and Genomic Medicine; The Methodist Hospital, Houston, TX, USA.
2MD, PhD, Medical Director, Surgical Pathology; Department of Pathology and Genomic Medicine (Houston, TX, USA), Senior Member; The Methodist Hospital Physician Organization (Houston, TX, USA), Professor of Pathology and Laboratory Medicine; The Methodist Hospital Research Institute; Weill Cornell Medical College of Cornell University, Houston, TX, USA. 3MD, Deputy Chief of Pathology; Department of Pathology and Genomic Medicine (Houston, TX, USA). Senior Member; The Methodist Hospital Physician Organization (Houston, TX, USA). Professor of Pathology and Laboratory Medicine; The Methodist Hospital Research Institute; Weill Cornell Medical College of Cornell University, Houston, TX, USA. | ||||||
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| How to cite this article: |
| Jerz JL, Ro JY, Ayala AG. Refractory bilateral scrotal ulcers with numerous IgG4-positive plasma cells: Another skin location for IgG4-related sclerosing disease. International Journal of Case Reports and Images 2014;5(2):131–134. |
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Abstract
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Introduction:
Immunoglobulin G4-related sclerosing disease (IgG4-RSD) is an emerging diagnosis characterized by mass-forming inflammation which responds to corticosteroids. Cases have been reported in virtually every organ, however, skin cases are rare and tend to involve the head and neck region.
Case Report: An 85-year-old male presented with painful, bilateral, non-healing scrotal ulcers refractory to treatment with antibiotics and creams. On examination, he was found to have multiple large, purulent, stage three scrotal ulcers. He was admitted for administration of broad-spectrum antibiotics, but the cultures of the ulcer discharge grew normal, non-pathogenic skin flora. Dermatology was consulted for evaluation of other non-infectious etiologies for the ulcers’ failure to heal. A shave biopsy showed 50% IgG4-positive plasma cells, and light-chain in situ hybridization studies demonstrated polyclonality. The patient’s serum IgG4 was elevated to 115 mg/dL (reference range 7–89 mg/dL), and he had an unclear remote history of Whipple surgery. The patient was discharged home with topical clobetasol (a synthetic corticosteroid) and required no follow-up for the ulcers. Conclusion: IgG4-RSD includes a variety of disease entities and may manifest as diffuse or solitary inflammatory mass lesions. Older males are affected most often overall. The diagnosis is made on histopathology with >40% IgG4-positive plasma cells, fibrosclerosis, and obliterative phlebitis, regardless of organ. Elevated serum levels of IgG4 are markers of disease, but not required for diagnosis. Given the patient’s histologic findings and laboratory results, this case is likely the first report of IgG4-RSD in scrotal skin. | |
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Keywords:
IgG4, Sclerosing disease, Pseudolymphoma, Corticosteroids
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Author Contributions
Jaclyn L Jerz – Conception and design, Acquisition of data, Analysis and interpretation of data, Drafting the article, Critical revision of the article, Final approval of the version to be published Jae Y Ro – Conception and design, Acquisition of data, Analysis and interpretation of data, Drafting the article, Critical revision of the article, Final approval of the version to be published Alberto G Ayala – Conception and design, Acquisition of data, Analysis and interpretation of data, Drafting the article, Critical revision of the article, Final approval of the version to be published |
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Guarantor of submission
The corresponding author is the guarantor of submission. |
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Source of support
None |
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Conflict of interest
Authors declare no conflict of interest. |
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Copyright
© Jaclyn L Jerz et al. 2014; This article is distributed the terms of Creative Commons Attribution License which permits unrestricted use, distribution and reproduction in any means provided the original authors and original publisher are properly credited. (Please see Copyright Policy for more information.) |
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