09 August 2021: Articles
Recurrence of Disseminated Presenting as Spondylodiscitis and Epidural Abscess in a Patient with Acquired Immune Deficiency Syndrome (AIDS)
Unusual clinical course, Challenging differential diagnosisMichael S. Wang12ABEF*, Nicholas M. Frazier12BE, Rhonda Griffiths1BE, Christian W. Sikorski34BE, Richard W. Douce12BEF
Am J Case Rep 2021; 22:e931595
BACKGROUND: Mycobacterium avium intracellulare complex (MAI) is a member of the non-tuberculous mycobacteria family, which can cause both pulmonary and non-pulmonary disease. In patients with advanced HIV, it is known to cause disseminated disease. We present a case of a 65-year-old man who has sex with men (MSM) with AIDS, found to have spondylodiscitis and an epidural abscess, who had recently completed treatment for disseminated MAI.
CASE REPORT: The patient was a 65-year-old with AIDS secondary to HIV and a prior history of disseminated MAI, who presented with severe back pain. Upon presentation to the hospital, an MRI was performed, which was suggestive of spondylodiscitis and an epidural abscess. He was taken to surgery for a minimally invasive T12-L1 laminectomy and evacuation of the epidural abscess. Both traditional cultures and acid-fast bacillus (AFB) cultures were negative. Due to worsening pain, he was taken back to surgery for a repeat debridement and biopsy. Repeat cultures were positive for MAI. He was started on rifabutin, ethambutol, azithromycin, and moxifloxacin. Moxifloxacin was subsequently discontinued. He has had problems tolerating the treatment regimen, but is planned to complete an 18-24-month course.
CONCLUSIONS: For patients with AIDS who have a diagnosis of spondylodiscitis and an epidural abscess, an opportunistic infection such as MAI should be considered. A repeat biopsy should be considered if suspicion is still high, even despite initially negative cultures. Treatment regimens should be prolonged, despite difficulty with medication compliance.
Keywords: Epidural Abscess, HIV, Mycobacterium Infections, Nontuberculous, Acquired Immunodeficiency Syndrome, Aged, Discitis, Homosexuality, Male, Mycobacterium avium Complex, Mycobacterium avium-intracellulare Infection, Sexual and Gender Minorities
Disseminated MAI is a clinical diagnosis which is distinctive from pulmonary MAI, both in terms of risk factors and clinical presentation [1,2]. Pulmonary MAI often occurs in both immunocompetent and immunosuppressed patients, and structural lung disease is a risk factor . Disseminated MAI tends to occur in those with profound immunosuppression . Patients with AIDS, particularly those with a CD 4 <50 cells/uL, are at high risk for disseminated MAI . In such patients with advanced immunosuppression, MAI disease often is a disseminated, multi-organism infection . Disseminated non-tuberculous mycobacteria (NTM), including MAI, have been documented to cause vertebral osteomyelitis, particularly in immunocompromised patients [3–5]. Here, we present a case of a 65-year-old MSM with a history of AIDS who was found to have spondylodiscitis and an epidural abscess secondary to recurrent and disseminated MAI.
We presumed the possibility of tuberculosis or resistant MAI, and thus patient was started on isoniazid 300 mg daily, azithromycin 500 mg daily, ethambutol 800 mg daily, moxifloxacin 400 mg daily, and rifabutin 300 mg daily, pending antimicrobial sensitivities, and was continued on previously prescribed dapsone and his antiviral regimen. An AFB culture was positive for MAI, but the isolate was deemed non-viable for susceptibility testing. Upon identification of MAI, the isoniazid was discontinued. The patient initially did not tolerate this initial regimen, developing refractory nausea and vomiting, believed to be secondary to rifabutin, which was discontinued and subsequently resulted in symptom resolution. Within a few more weeks, he again developed refractory nausea and vomiting, at which time moxifloxacin was stopped, again with resolution of symptoms. Out of the desire to have the patient on a 3-drug regimen, 1 month later rifabutin was resumed at a lower dose and to date this regimen has been tolerated well. He has continued on azithromycin 500 mg daily, ethambutol 800 mg daily, and rifabutin 150 mg 3 times per week.
MAI is found in water and soil environments, with water being the main source of infection due to innate chloride resistance . The pathogenesis of MAI in AIDS patients is believed to be secondary to gastrointestinal colonization and subsequent invasion across the mucosa and submucosa . The present patient had a history of disseminated MAI, likely secondary to his immunosuppression from AIDS. Macrolide-based treatment courses for at least 12 months have been successful in several case series, particularly with recovery of CD4 cell counts [8–10]. Although rare, relapses have been documented, including one case with osteomyelitis . Despite being treated for 12 months, we believe he likely had residual infection which presented as spondylodiscitis and an epidural abscess. Complicating his course was an inability of his CD4 cell count to consistently rise >200 cells/uL.
Mycobacterium avium complex has been documented to cause spondylodiscitis, both in HIV and non-HIV patients [3–5]. Among mycobacteria, Mycobacterium tuberculosis has been well-documented to cause spondylodiscitis . Other cases of NTM include M. xenopi, M. abscessus, M. chelonae, M. fortuitum, M. kansasii, M. siniae, and M. flavescens . The majority of these cases were either immunocompromised or on immunosuppressive medication, including steroids [3–5]. One series noted that 14.5% of patients with NTM vertebral osteomyelitis were positive for HIV . Immune reconstitution may contribute to diagnosis of spondylodiscitis, as patients were diagnosed an average of 12.5 months from initiation of antiretroviral therapy . We speculate that immune reconstitution may have contributed to his presentation, given that his CD4 had risen steadily over the previous 6 months. Complicating factors for our patient included intermittent compliance with his antiretroviral therapy. An epidural abscess is an even rarer complication of disseminated MAI . It has been previously documented in M. tuberculosis .
Diagnosing spondylosicitis and epidural abscess frequently requires a surgical biopsy, particularly if blood cultures are negative for Staphylococcus aureus, Staphylococcus lugdunensis, or Brucella . In the case of this patient, his blood cultures, which were drawn after antibiotics were initiated, were negative. Previous cases of MAI spondylosicitis frequently required a surgical biopsy, with AFB cultures being the primary diagnostic test [3,4]. Given the high suspicion for an opportunistic pathogen, the patient was taken to the operating room for a biopsy, including AFB and fungal cultures, and drainage of the epidural abscess. Due to negative cultures and the patient’s lack of improvement, he was taken back to surgery for a second biopsy and drainage of the abscess. A few cases in the medical literature required 2 biopsies, but at least 1 case was secondary to a lack of initial AFB cultures due to low suspicion .
In terms of treatment, the Infectious Diseases Society of America treatment guidelines recommend 3-drug therapy for pulmonary disease, with a strong emphasis on a macrolide and ethambutol . Similarly, treatment for disseminated MAI, which this likely represents, often may include rifampin, rifabutin, clofazimine, fluoroquinolones, and aminoglycosides [2,6]. This patient was previously treated with 12 months of therapy for MAI. Previously successful regimens for vertebral osteomyelitis were similar to those prescribed for disseminated MAI [3,4]. NTM spondylodiscitis patients improved in approximately 80% of cases . A case review of 16 non-HIV vertebral osteomyelitis secondary to MAI found that 68.8% of patients improved with therapy , with a mean duration of 16.8 months.
Our patient had difficulty tolerating rifabutin and, subsequently, moxifloxacin. We did not use rifampin due to potential drug-drug interactions with the protease inhibitor. As previously noted, he has been on darunavir-cobicistat, dolutegravir, and lamivudine for antiretroviral therapy. After considerable encouragement, he resumed the rifabutin and has mostly been compliant, but has had lapses in his MAI therapy. Based on our previous literature review , we opted to continue him on 3-drug therapy for his spondylosicitis and epidural abscess. Given that he relapsed despite 12 months of therapy and intermittent compliance, we will aim to treat him for at least 18–24 months.
Our patient with AIDS and disseminated MAI presented with spondylosicitis and an epidural abscess and required a second biopsy procedure to confirm the diagnosis. He improved with irrigation and drainage of the abscess and continues to do well on 3-drug therapy with azithromycin, rifabutin, and ethambutol.
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