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

Abstract

Background Inflammatory myofibroblastic tumor (IMT) is a rare disease that mostly occurs in younger people and is located in the lungs in the general population. We report a rare case of adrenal IMT in a patient with HIV infection, which is believed to be the first of its kind worldwide. Case presentation We present a rare case of a 44-year-old man with HIV infection who was diagnosed with adrenal IMT. The patient refused regular highly active antiretroviral therapy 13 years ago until he was admitted to hospital after an adrenal mass was found. The patient underwent successful computed-tomography-guided needle biopsy, and pathological analysis showed fibroblastic–myofibroblastic proliferation with inflammatory infiltration, which confirmed a diagnosis of IMT. We failed to perform complete resection of the tumor because of its diffuse invasion. The patient was complicated with severe multiple pulmonary infections postoperatively because of immunodeficiency, which eventually caused his death 2 months later. Conclusion Differential diagnosis of IMT is difficult, and tumor biopsy is an essential means of diagnosis. Surgical resection is preferred for both adrenal and HIV-related IMTs. Conservative treatment should be considered when there are technical difficulties with complete resection, and most patients have achieved good outcomes. However, more cases and longer follow-up are warranted to confirm long-term outcomes of HIV-related IMT.

Inflammatory myofibroblastic tumor from molecular diagnostics to current treatment

“…It also presents a strong predilection for visceral organs and deep soft tissues of the abdomen, pelvis, retroperitoneum [8][9][10][11], head, and neck [12,13], but any part of the human body may be affected, including somatic soft tissues, bone, extremities, larynx, or even central nervous system [9,14]. Some rare locations include the esophagus [15], the pericardium [12], the heart [7], the spinal meninges [15], and the adrenal glands [16,17]. A case describing the simultaneous location of IMT lateral to the ascending aorta near the root and inside the atrial septum has been reported [18].…”

“…At diagnosis, pulmonary IMT is associated with upper respiratory infection or pneumonia in 30% of patients [ 39 , 40 ]. Tumors located in the abdomen and pelvis may cause symptoms that vary according to the location: nausea, vomiting [ 41 , 42 ], dysphagia [ 43 ], reflux [ 36 , 44 ], melaena [ 45 ], abdominal or back pain [ 16 , 42 ], hematuria [ 46 ], menorrhagia [ 47 ] or even gastrointestinal obstruction [ 48 ]. Tumors range from 1 to >20 cm in maximum dimension [ 10 , 26 , 30 , 37 , 41 , 49 – 51 ], with a mean size of 4–8 cm [ 11 , 27 , 52 , 53 ].…”

Inflammatory Myofibroblastic Tumor in the Adrenal Gland: A Case Report

“…Distant metastases are rare but mainly occur in the lungs and brain followed by liver and bone. If complete resection is not performed, 10–25% of cases will have local recurrence, and <5% will have distant metastasis, which may occur between 1 and 9 years after initial presentation [5]. IMTs of the adrenal gland are usually discovered incidentally, but it may also be accompanied by fever of unknown origin and abdominal pain.…”