Acute bilateral parotitis caused by Mycobacterium scrofulaceum: immune reconstitution disease in a patient with AIDS
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1 Department of Infectious Diseases, St George’s Hospital Medical School, London SW17 0RE, UK
2 Department of Infectious and Tropical Diseases, London School of Hygiene and Tropical Medicine, London, UK
Keywords: HIV-1; antiretroviral treatment; immune reconstitution; Mycobacterium; parotid gland
Immune reconstitution disease (IRD) among HIV infected patients is an adverse consequence of restoration of immune responses during the initial months of antiretroviral treatment (ART).1 Previously, subclinical infections are "unmasked" or pre-existing opportunistic infections clinically deteriorate. Here we describe an unusual case in which a patient developed acute bilateral parotid enlargement as a result of IRD associated with Mycobacterium scrofulaceum infection.
A 66 year old West African man was investigated for dysphagia, weight loss, and fatigue. Oesophageal candidiasis and HIV-1 infection were diagnosed with a blood CD4 lymphocyte count of 6 cells x106/l and a plasma viral load of 416 566 RNA copies/ml. Further investigations, including chest radiography, sputum examination, mycobacterial blood cultures, bone marrow examination and culture, and abdominal ultrasonography, were normal. Following treatment of oesophageal candidiasis, the patient started ART with zidovudine, lamivudine, and efavirenz.
Eight weeks later, the patient developed acute swelling posterior to the angles of the jaw bilaterally, causing discomfort on mouth opening. He did not have xerostomia or other local or systemic symptoms. On examination, firm smooth masses were found, more marked on the right than the left, arising from the behind the angles of the jaw and extending inferiorly into the neck (fig 1). The patient was afebrile and no lymphadenopathy was palpable elsewhere. Clinical examination was otherwise normal, including the oropharynx and seventh cranial nerve function.
Ultrasound examination confirmed that both parotid glands were diffusely enlarged and showed increased vascularity; intraparotid lymphadenopathy was not evident. However, several cervical lymph nodes were also unilaterally enlarged on the right (fig 1). Blood investigations revealed no inflammatory response and normal serum concentrations of angiotensin converting enzyme, calcium and amylase and autoantibodies. A chest radiograph and abdominal ultrasound were normal. No pathogens were identified in sputum or in blood cultures for bacteria and mycobacteria. Immunoglobulin G to mumps virus was detectable in serum, indicating previous infection. A fine needle aspirate (FNA) of the right parotid was paucicellular, precluding a cytological diagnosis. The patient’s blood CD4 count had rapidly increased to 80 cells x106/l and the viral load was undetectable. A presumptive diagnosis of IRD was made, although any underlying infection was unknown.
Two weeks later, the right sided lymph nodes had enlarged further. M scrofulaceum was cultured from the original FNA. Treatment with rifabutin and clarithromycin was started and ART was continued. The right sided lymph nodes became fluctuant and discharged pus, which contained acid fast bacilli but was culture negative. The parotitis and lymphadenitis subsequently resolved over several weeks.
M scrofulaceum typically causes cervical lymphadenopathy in children and is a rare cause of disease in patients with HIV/AIDS2; parotid disease has not previously been reported. Mycobacteria are the organisms most frequently reported to underlie IRD, which commonly presents with acute lymphadenitis or deterioration of pulmonary disease.1 However, this is the first report of mycobacteria associated IRD presenting with parotid disease. The differential diagnosis of parotid disease in patients with HIV infection is broad, and includes infections, malignancies, benign lymphoepithelial cysts, diffuse infiltrative lymphocytosis syndrome and Sj?gren’s syndrome.3,4 Clinicians should also be aware that acute parotid enlargement may also be the result of IRD.
ACKNOWLEDGEMENTS
SDL is funded by the Wellcome Trust, UK.
CONTRIBUTORS
SDL wrote the manuscript; all authors were involved in the clinical care of the patient, data collection, and input to the final manuscript.
FOOTNOTES
Conflicts of interest: The authors have no conflicts of interest.
References
Shelburne SA III, Hamill RJ. The immune reconstitution inflammatory syndrome. AIDS Rev 2003;5:67–79.
Sanders JW, Walsh AD, Snider RL, et al. Disseminated Mycobacterium scrofulaceum infection: a potentially treatable complication of AIDS. Clin Infect Dis 1995;20:549.
Seddon BM, Padley SP, Gazzard BG. Differential diagnosis of parotid masses in HIV positive men: report of five cases and review. Int J STD AIDS 1996;7:224–7.
Kazi S, Cohen PR, Williams F, et al. The diffuse infiltrative lymphocytosis syndrome. Clinical and immunogenetic features in 35 patients. AIDS 1996;10:385–91.(S D Lawn, A Checkley and )
2 Department of Infectious and Tropical Diseases, London School of Hygiene and Tropical Medicine, London, UK
Keywords: HIV-1; antiretroviral treatment; immune reconstitution; Mycobacterium; parotid gland
Immune reconstitution disease (IRD) among HIV infected patients is an adverse consequence of restoration of immune responses during the initial months of antiretroviral treatment (ART).1 Previously, subclinical infections are "unmasked" or pre-existing opportunistic infections clinically deteriorate. Here we describe an unusual case in which a patient developed acute bilateral parotid enlargement as a result of IRD associated with Mycobacterium scrofulaceum infection.
A 66 year old West African man was investigated for dysphagia, weight loss, and fatigue. Oesophageal candidiasis and HIV-1 infection were diagnosed with a blood CD4 lymphocyte count of 6 cells x106/l and a plasma viral load of 416 566 RNA copies/ml. Further investigations, including chest radiography, sputum examination, mycobacterial blood cultures, bone marrow examination and culture, and abdominal ultrasonography, were normal. Following treatment of oesophageal candidiasis, the patient started ART with zidovudine, lamivudine, and efavirenz.
Eight weeks later, the patient developed acute swelling posterior to the angles of the jaw bilaterally, causing discomfort on mouth opening. He did not have xerostomia or other local or systemic symptoms. On examination, firm smooth masses were found, more marked on the right than the left, arising from the behind the angles of the jaw and extending inferiorly into the neck (fig 1). The patient was afebrile and no lymphadenopathy was palpable elsewhere. Clinical examination was otherwise normal, including the oropharynx and seventh cranial nerve function.
Ultrasound examination confirmed that both parotid glands were diffusely enlarged and showed increased vascularity; intraparotid lymphadenopathy was not evident. However, several cervical lymph nodes were also unilaterally enlarged on the right (fig 1). Blood investigations revealed no inflammatory response and normal serum concentrations of angiotensin converting enzyme, calcium and amylase and autoantibodies. A chest radiograph and abdominal ultrasound were normal. No pathogens were identified in sputum or in blood cultures for bacteria and mycobacteria. Immunoglobulin G to mumps virus was detectable in serum, indicating previous infection. A fine needle aspirate (FNA) of the right parotid was paucicellular, precluding a cytological diagnosis. The patient’s blood CD4 count had rapidly increased to 80 cells x106/l and the viral load was undetectable. A presumptive diagnosis of IRD was made, although any underlying infection was unknown.
Two weeks later, the right sided lymph nodes had enlarged further. M scrofulaceum was cultured from the original FNA. Treatment with rifabutin and clarithromycin was started and ART was continued. The right sided lymph nodes became fluctuant and discharged pus, which contained acid fast bacilli but was culture negative. The parotitis and lymphadenitis subsequently resolved over several weeks.
M scrofulaceum typically causes cervical lymphadenopathy in children and is a rare cause of disease in patients with HIV/AIDS2; parotid disease has not previously been reported. Mycobacteria are the organisms most frequently reported to underlie IRD, which commonly presents with acute lymphadenitis or deterioration of pulmonary disease.1 However, this is the first report of mycobacteria associated IRD presenting with parotid disease. The differential diagnosis of parotid disease in patients with HIV infection is broad, and includes infections, malignancies, benign lymphoepithelial cysts, diffuse infiltrative lymphocytosis syndrome and Sj?gren’s syndrome.3,4 Clinicians should also be aware that acute parotid enlargement may also be the result of IRD.
ACKNOWLEDGEMENTS
SDL is funded by the Wellcome Trust, UK.
CONTRIBUTORS
SDL wrote the manuscript; all authors were involved in the clinical care of the patient, data collection, and input to the final manuscript.
FOOTNOTES
Conflicts of interest: The authors have no conflicts of interest.
References
Shelburne SA III, Hamill RJ. The immune reconstitution inflammatory syndrome. AIDS Rev 2003;5:67–79.
Sanders JW, Walsh AD, Snider RL, et al. Disseminated Mycobacterium scrofulaceum infection: a potentially treatable complication of AIDS. Clin Infect Dis 1995;20:549.
Seddon BM, Padley SP, Gazzard BG. Differential diagnosis of parotid masses in HIV positive men: report of five cases and review. Int J STD AIDS 1996;7:224–7.
Kazi S, Cohen PR, Williams F, et al. The diffuse infiltrative lymphocytosis syndrome. Clinical and immunogenetic features in 35 patients. AIDS 1996;10:385–91.(S D Lawn, A Checkley and )