The most common site of IMT is lung according to former studies,while adrenal gland is unusual anatomical location .The patient merely presented with back pain accompanied by no other manifestations such as fever, palpable mass, and emaciation. In addition, CT showed bilateral masses with no specifically enhanced images. Above all, the patient had a 13-year history of HIV-infection with no treatments, leading to collapsed immunal system. In light of the above aspects, AIDS-defining cancers were given priority in differential diagnosis such as lymphoma and Kaposi’s sarcoma. CT-guided biopsy of the lesion was necessary to make a diagnosis.
Pathological analysis documented a proliferation of spindle cells and an infiltration of plasma cells, lymphocytes and neutrophils in accordance with features reported in former studies [10, 11]. Immunohistochemical study showed that ALK1 was negative, representing less aggressive pathology and less likely to recur . A study involved 84 cases suggested a 25% increasement of recurrence rate for ALK1-positive IMT specimens versus those of ALK1 negative . However intraoperative findings showed more aggressive pathological features in our case, which was in inconsistent with the predictive effect of ALK. This contradiction might be caused by HIV infection.
Completely surgical resection is the main treatment for most IMTs, and mass biopsy should be recommended to avoid organ resection for certain patients, when it is difficult to differentiate from primary organ malignancy. A retrospective study including 22 IMTs of urinary system documented good outcomes with no recurrence or metastases in a median follow-up duration of 6.1 years, who were treated with completely mass resection or radical organ resection. If incomplete resection occurs, adjuvant therapy including steroid therapy, antibiotics, radiotherapy, chemotherapy or carbon dioxide laser should be attempted. Recent study showed that ALK inhibitors appeared to be beneficial to IMT as adjuvent therapy,which had been approved efficacy for ALK-positive non–small cell lung cancer . In a multicenter prospective study, 12 patients of IMTS achieved an objective response of 50%, who accepted adjuvent therapy of ALK inhibitor (crizotinib), and ALK inhibitor regarded as a kind of targeted therapy seemed to be quitely effective in treating IMTs with incomplete resection . We did not succeed in mass resection because of undefined margine and extensive invasion of the tumor in this case, that might be caused by immunodeficiency.We did not recommend any adjuvent therapies as well, because the patient was complicated with uncontrolled pulmonary infection that might be caused by endotracheal intubation in operation.
IMT was used to considering as benign tumor since it was firstly reported in lung in 1939 . Most of IMTs had good outcomes according to the former studies, that relied on many factors such as demographics, comorbidities, oncologic features, expression of ALK, and degree of surgical completion[4, 14]. Nowadays, pathologists demonstrate IMT’s aggressive features of pathology in certain cases, suggesting its malignant potential [18, 19]. Neverthless, the outcome of IMT is uncertain when it comes to HIV infection due to rare case reports of HIV-related IMTs. As far as we know, only 1 case had previously been reported in Romania, who didn’t undergo surgical resection but received broad-spectrum antibiotics, antiviral and antifungal treatment, and the case had good evolution without any recurrence during 3-year follow-up. In our case, the patient finally died of AIDS complications post to unsuccessful surgery withthin 2 months, that made it impossible to try adjuvent therapies and track the natural course of tumor.
Our case and Romania case were featured by collapsed immunity caused by HIV. It is reasonable to speculate that there may be a possible correlation between HIV and IMT. More studies of HIV-related IMTs are necessary to confirm that it is accidental or not.