Inflammatory pseudotumor associated with HSV infection of rectal vascular endothelium in a patient with HIV: a case report and literature review

Abstract

Background: Herpes simplex virus (HSV) typically infects oral or anogenital squamous epithelium and causes blisters and ulcerations. Here we reported an unusual case of HSV induced exuberant rectal inflammatory pseudotumor with vascular endothelial involvement. Case presentations: A 52-year old man with HIV presented with abdominal pain, rectal drainage and constipation. Proctoscopy and CT scans revealed an 8 × 5 × 4 cm circumferential, mid-lower rectal mass that was concerning for malignancy. PET-CT showed mild to moderate FDG uptake of the rectal mass. Repeated biopsies showed exuberant lymphoplasmacytic inflammation with rich eosinophils and necrosis in the submucosa and scattered single or multinucleated viral inclusions in vascular endothelial cells that were positive for HSV by immunostains. There was no evidence of malignancy on histology or by immunostains. The patient started valacyclovir for three weeks and symptoms resolved after the antiviral therapy. Follow-up CT and sigmoidoscopy with biopsy revealed no rectal mass or drainable collection. Conclusions: HSV may present as proctitis with exuberant inflammatory response and mass-like lesion, and damages vascular endothelial cells in patients with HIV. The HSV-associated mass-like lesion can be effectively treated by 3-week valacyclovir.

HIV-related bilateral inflammatory myofibroblastic tumors of the adrenal gland: A case report and literature review

“…Nevertheless, the outcome of IMT is uncertain when it is related to HIV infection because there are only a few relevant case reports. To our knowledge, only 10 cases have been reported globally; most of which seemed to have good outcomes after follow-up of 3 months to 1 year [9][10][11][12][13][14][15][16][17]. In our case, the patient died of AIDS-related complications within 2 months of unsuccessful surgery that made it impossible to try adjuvant therapies and track the natural course of the tumor.…”

“…Human papillomavirus and Helicobacter pylori have also been con rmed in IMT tissues, but the speci c mechanism remains to be fully investigated, which may be relevant to the cytokine release and B-lymphocyte differentiation caused by infection [11,20]. The etiology of HIV-related IMT should also include the above mechanism, given that immunostaining in recent cases has demonstrated herpes simplex virus infection in rectal IMT [17]. Some experts believe that the occurrence of HIV-related IMT has a potential relationship with immune reconstitution in ammatory syndrome (IRIS), which is characterized by a paradoxical clinical worsening under the background of an improving immune system.…”

“…We did not recommend any adjuvant therapy because of the complication of uncontrolled pulmonary infection that might have been caused by endotracheal intubation during anesthesia. In previous studies of HIV-related IMT, some cases underwent successful mass resection [11][12][13][14]16], while the remaining cases had the same conservative treatment as the general population [9,15,17].…”

“…However, the increased incidence of non-AIDS-de ning cancers has been accompanied by improved life expectancy because of the advent of highly active antiretroviral therapy (HAART) worldwide [7]. IMT is a type of non-AIDS-de ning cancer that has only been reported in a few patients with HIV infection [8][9][10][11][12][13][14][15][16][17]. We present what is believed to be the rst case worldwide of HIV-related adrenal IMT, and a review of the literature on HIV-related IMTs and adrenal IMTs.…”

“…F female, IMT in ammatory myo broblastic tumor, M male, NA not available, NR no recurrence, NP no progression Previous studies have shown that the most common site of IMT is the lungs, while the adrenal gland is an unusual location[18]. However, only one case of HIV-related IMT occurred in the lungs, and its distribution was scattered over different sites, which is obviously different from the general population[8][9][10][11][12][13][14][15][16][17]. Our patient presented with back pain accompanied by other manifestations such as fever, fatigue, and emaciation, which was similar to former reports of adrenal IMT[19][20][21][22][23][24][25][26][27].…”