Genital pyoderma gangrenosum: Report of two cases and published work review of Japanese cases

Satoh, Masataka; Yamamoto, Toshiyuki

doi:10.1111/1346-8138.12252

Cited by 40 publications

(31 citation statements)

References 6 publications

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“…Fewer than 20 cases have been reported affecting the penis 11–22. PG can be associated with systemic diseases such as Crohn's disease, rheumatoid arthritis and haematological malignancies, but it also complicates trauma and surgical procedures 11–15 22. Crohn's disease is a chronic relapsing, granulomatous and inflammatory bowel disorder 11 23.…”

Section: Discussionmentioning

confidence: 99%

Necrotizing granulomatous inflammation of the glans penis

Christodoulidou¹,

Bunker

Trevisan

et al. 2016

BMJ Case Reports

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We describe the case of a 73-year-old man who presented with a 10-month history of an ulcerating lesion on the glans penis. Initially this was thought to be an invasive squamous cell carcinoma but a biopsy showed histological features consistent with necrotizing granulomatous inflammation. Extensive serological, immunological and microbiological tests only showed a positive antinuclear and perinuclear antineutrophil cytoplasmic antibodies indicating a possible autoimmune aetiology but an underlying systemic cause was not identified. Treatment with oral corticosteroids limited the inflammatory process but due to the gross destruction of the glans penis, he still required a glansectomy and split-skin graft reconstruction from which he recovered well. Although this patient ultimately required surgery for this rare presentation, this case highlights the differential diagnosis of penile ulceration (that transcends neoplasia) and the importance of performing and interpreting penile biopsies before undertaking potentially mutilating definitive surgery.

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Section: Discussionmentioning

confidence: 99%

Necrotizing granulomatous inflammation of the glans penis

Christodoulidou¹,

Bunker

Trevisan

et al. 2016

BMJ Case Reports

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“…PG is an ulcerative skin disorder showing characteristic non-infectious ulcers and affects the lower extremities in approximately 70 % of cases. PG is commonly associated with systemic diseases such as inflammatory bowel disease, rheumatoid arthritis and hematological malignancies [20]. Ulcerations of the skin and mucosal membranes are a common feature of some primary immune deficiencies such as autoinflammatory diseases.…”

Section: Discussionmentioning

confidence: 99%

Atypical Severe Combined Immunodeficiency Caused by a Novel Homozygous Mutation In Rag1 Gene in a Girl who Presented with Pyoderma Gangrenosum: A Case Report and Literature Review

et al. 2014

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Severe combined immunodeficiency (SCID) is a heterogeneous group of inherited defects involving the development of T- and/or B-lymphocytes. We report a female with atypical severe combined immunodeficiency caused by a novel homozygous mutation at cDNA position 2290 (c.2290C > T) in exon 2 of the RAG1 gene. The patient presented with bronchopneumonia, pyoderma gangrenosum (PG), pancytopenia and splenomegaly. She presented to us with pancytopenia and splenomegaly at the age of 11. Her condition was complicated by PG on left lower ankle at the age of 12. She experienced bronchopneumonia at the age of 15. She was diagnosed with RAG1 deficiency at the age of 16. Her immunological presentation included leucopenia and diminished number of B cells.

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“…Due to the clinicopathological features, consideration of a variant of pyoderma gangrenosum was proposed, but the early age onset, unusual localization, histology of vasculitis, rapid favourable treatment response, benign lenient disease course in the absence of underlying diseases are different to the manifestations of classical pyoderma gangrenosum …”

Section: Juvenile Gangrenous Vasculitis Of the Scrotummentioning

confidence: 99%

Lipschütz genital ulcer revisited: is juvenile gangrenous vasculitis of the scrotum the male counterpart?

Chen

Plewig

2019

Acad Dermatol Venereol

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Since its first description as ulcus vulvae acutum by Benjamin Lipschütz in 1912, the etiopathogenesis of this peculiar genital ulcer remains incompletely understood. In his original description, two different types of genital ulcers were observed and proposed, which were not precisely defined and distinguished in most subsequent reports. The first type is characterized by acute excruciating genital ulcers of first‐time onset with self‐limited non‐recurrent course in association with gravely symptomatic systemic infections, in which a primary Epstein–Barr virus (EBV) infection is later identified to be probably the most common aetiology. The second type of ulcer usually refers to little painful ulcers of unknown etiopathogenesis in the absence of fever or chills and with a slow torpid progression and recurrent nature. Differentiation from idiopathic aphthous ulcers is unclear. The changes of the cervicovaginal microbiota and microbiome in diseased state deserve further clarification. Acute genital ulcers associated with primary EBV infection in women have drawn attention since 1970s, while the corresponding penile ulcers in men were already known in 1950s. First presented in 1973, juvenile gangrenous vasculitis of the scrotum with an acute painful scrotal ulcer preceded by symptomatic pharyngeal infections can be considered as the male counterpart of ulcus vulvae acutum, and the future clinical survey should include primary EBV infection.

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Genital pyoderma gangrenosum: Report of two cases and published work review of Japanese cases

Cited by 40 publications

References 6 publications

Necrotizing granulomatous inflammation of the glans penis

Necrotizing granulomatous inflammation of the glans penis

Atypical Severe Combined Immunodeficiency Caused by a Novel Homozygous Mutation In Rag1 Gene in a Girl who Presented with Pyoderma Gangrenosum: A Case Report and Literature Review

Lipschütz genital ulcer revisited: is juvenile gangrenous vasculitis of the scrotum the male counterpart?

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