Actinomycosis is a rare disease caused by bacteria from the genus Actinomyces spp., which colonizes the gastrointestinal, respiratory, and genitourinary tract as part of normal microbiota. However, chronic injury of a colonized area may lead to disease. [1, 3, 10] Cases have been described in PLWH and most reports describe lymphocyte T CD4 < 200 cell/mL. [4–7] Furthermore, reports usually highlight underlying conditions which accompany the diagnosis of esophageal actinomycosis, such as esophageal candidiasis, cytomegalovirus esophagitis or neoplasia.
Esophageal actinomycosis does not usually involve the distal third of the esophagus due to its distance from the oropharynx, [11] but can present as chronic esophageal ulceration [12, 13] or even mimicking esophageal carcinoma, [14–17] as in our case. Neurological dysphagia leading to aspiration constitutes a further risk factor that has been reported in some cases and is associated with distal esophageal compromise. [10, 11, 17] In the case we describe, probably both the digestive mucosa MALT-lymphoma-associated changes as well as HIV-associated immunosuppression led to the development of esophageal actinomycosis.
Clinical manifestations of esophageal actinomycosis range from dyspepsia and dysphagia to weight loss and wasting, mimicking the clinical presentation of a neoplasm. [18] While our patient had dysphagia, but was able to eat and drink, and had a CD4 + T lymphocyte count greater than 200 cells/ml, it is possible that the actinomycosis alone did not account for the wasting syndrome and therefore, further studies to explain emaciation were justified. Even though HIV infection could justify the weight loss, the not so low CD4 + T lymphocyte count (314 cells/ml) strongly leads us to believe that this was the result of underlying neoplasm.
Actinomycosis shows a good clinical response to long courses of amoxicillin, penicillin G, doxycycline, or metronidazole, ranging from 6 to 12 months. [7] Such is the case of this patient, whose mass resolved leaving only a well healing scar without any evidence of strictures and resolution of all initial symptoms.
This patient presented HIV-associated immunosuppression [1, 4] as the only risk factor for invasive actinomycosis. Nevertheless, it has been reported that PLWH may present actinomycosis with CD4 cell counts as high as 300 cells/mL. [6, 19, 20] Other small studies in patients with invasive actinomycosis showed that it is a surrogate marker of poor prognosis in this specific vulnerable hosts. [21] Despite our patient had an initial CD4 count of 314 cells/ml, due to immunosuppression stigma and presence of neoplasm, we considered him to be in an advanced stage of HIV.
Proper CART naive-treatment was started with the combine dose formulation of Tenofovir-DF/Lamivudine/Dolutegravir. After 12 weeks of treatment, he reached a viral load of 164 copies/ml. Even though recurrence of invasive actinomycosis is extremely rare, [22] presence of immunocompromised state such as untreated HIV infection; puts patients increased risk of recurrence especially if the CART is inconsistent. Our case did not present recurrent actinomycosis because there was clinical and structural resolution of esophageal ulcers. In the context of viral control with appearance of new symptoms, it’s reasonable to consider other opportunistic infections. The UDS showed one new gastric lesion compatible with unmasked non-Hodgkin lymphoma-associated IRIS after CART and actinomycosis treatment.
In conclusion, despite its unusual occurrence, esophageal actinomycosis in PLWH in an advanced stage should arise suspicion for further opportunistic conditions that overlap or hide the clinical manifestations. PLWH and actinomycosis should be strictly monitored until viral control is achieved, and antibiotic courses are completed.