Isolated Oral Histoplasmosis Presenting as Fever of Unknown Origin in a Portuguese Hemodialysis Patient

Abstract

The authors report a clinical case of an isolated oral histoplasmosis in a hemodialysis patient that presented with fever of unknown origin and had an unremarkable physical examination. During the investigation, a Gallium scan showed uptake in the oral cavity and soon after the oral cavity examination revealed a granulomatous lesion on the tooth 26. Histopathologic findings were compatible with histoplasmosis. The treatment regimen included liposomal amphotericin B followed by itraconazole consolidation therapy, and side effects did not occur. Both clinical evolution and outcome were favorable. Oral histoplasmosis in a non-immunosuppressed patient is extremely rare.

Histoplasmosis Diagnosed in Europe and Israel: A Case Report and Systematic Review of the Literature from 2005 to 2020

“…We confirmed that the latency period from the last time spent in an endemic area to the time of diagnosis can be extremely long, except in the case of travellers with acute pulmonary histoplasmosis: median latency is 66 months among PLWH, 6 years among immunocompetent expatriates/migrants, and between as short as 3 weeks [83] and as long as 42 years [78] among patients with OIC. As previously shown by epidemiological studies of endemic areas [152] and among travellers [8], a majority of our cases involved males (440/659 66.8%) [12,, although the male-to-female ratio was lower (2:1) than that observed in other studies (4:1) [152].…”

“…The countries reporting the most cases were France (8, 28.6%), Spain (6, 21.4%), and The Netherlands (5, 17.8%). Sixteen patients had progressive disseminated histoplasmosis (57.1%), which was revealed by a picture of haemophagocytic lymphohistiocytosis (HLH) in five cases [76,83,84,88,92]; two had intestinal histoplasmosis [89,91], one of whom was initially misdiagnosed as having Crohn's disease leading to HLH following treatment with infliximab [91]; the remaining ten had apparently localised disease with cerebral involvement [72], endocarditis [77], pyomyositis [63], or pulmonary [87], oral [78], laryngeal [80], liver [44], osteoarticular [89] or skin diseases [39]. Seven cases (25%) were characterised by ulcerated lips or the involvement of the oropharyngeal mucosa [70][71][72]74,81,84,88].…”

Tonsillar ulceration as manifestation of disseminated African histoplasmosis in an immunocompetent Portuguese host

“…There are several cases worldwide reported with oral histoplasmosis in immunocompetent patients [7], [9], [10], [11], [12], [13], [14], [15], [16], [17], [18], [19]. In Portugal, only one case was published about an oral histoplasmosis in a hemodialysis patient with persisted fever [9].…”

“…In Portugal, only one case was published about an oral histoplasmosis in a hemodialysis patient with persisted fever [9].…”

“…Variety duboisii has tropism for lymph nodes, skin, and bones and is classically associated with cutaneous lesions (nodules, ulcers) and osteolytic bone lesions, especially affecting the skull, ribs, and vertebrae [6]. The disseminated form is defined by the presence of an extra-pulmonary focus, particularly on those with risk factors: immunosuppression, transplantation, chronic renal disease, prolonged use of corticosteroids and acquired immune deficiency syndrome [7], [9]. One third of disseminated disease presented oropharyngeal involvement.…”