A 53-year-old gentleman, known diabetic of 8 years duration presented with a pain in both lower limbs of 15 days duration and weakness in the form of ascending quadriparesis involving proximal followed by distal group of muscles of 12 days duration. There were no cranial nerve and respiratory symptoms. There was history of malaise and low grade fever preceding this illness.\nOn examination at admission, vital signs were normal. General physical examination was normal. Higher mental functions and cranial nerve examination were normal. There was hypotonia in all four limbs with Medical research council (MRC) power of grade 4/5. The deep tendon reflexes were absent and he had bilateral flexor plantar response. Cerebrospinal fluid examination revealed albumino-cytological dissociation (Glucose 70 mg%, protein 88 mg%, no cells). Nerve conduction studies were suggestive of demyelinating polyradiculoneuropathy. He was detected to be HBs Ag positive and was seronegative for human immunodeficiency virus infection. Apart from elevated glycosylated hemoglobin A1C level of 7.8%, serum biochemical parameters including potassium levels were normal. He had neutrophilic leukocytosis (13,400/cmm, 88% neutrophils), blood cultures were sent and was empirically started on ceftriaxone. IV immunoglobulins of 0.40 g/kg/day were given for 5 days. During the course of the hospital stay, his blood sugars were kept under control with insulin.\nOn 2nd day of admission, patient developed bilateral facial weakness, power deteriorated to MRC grade 3/5. After 4 days of treatment, the patient developed respiratory distress requiring ventilatory support. Arterial blood gases report showed metabolic acidosis with raised lactate levels and hypoxia. Later, patient developed refractory septic shock and succumbed to death. Cultures grew B. pseudomallei and the diagnosis of melioidiosis presenting as GBS on a background of diabetes mellitus was considered.
A 26-year-old female with B. cenocepecia presented with a history of recurrent small joint pains in association with intermittent painful erythematous nodular rash on the lower legs. This came on at the onset of an infective exacerbation of her chest. Biopsy confirmed erythema nodosum. Other causes of erythema nodosum were excluded, and she did not respond to Non-steroidal anti-inflammatory medications. Her symptoms continue to relapse in association with infective exacerbations of her chest.\nOf the remaining patients (n = 3), one presented with an erythematous rash on the lower legs and another had a purpuric rash on the lower legs and both were diagnosed clinically as leucocytoclastic vasculitis by a dermatologist. The third patient presented with purpuric areas and blistering of the lower legs which was confirmed as leucocytoclastic vasculitis on biopsy.\nOverall, two patients had biopsy proven leucocytoclastic vasculitis, three were clinically diagnosed with vasculitis but did not have biopsy confirmation and two had biopsies, which revealed other pathologies (erythema nodosum and capillaritis), giving an overall prevalence of cutaneous vasculitis in patients with Burkholderia cepacia complex of 16.7%. Out of the five patients with vasculitis, three had a raised ESR/CRP ratio.\nThere was no evidence of renal involvement in any patient – all had a negative urine dipstick and normal serum creatinine. The antinuclear antibody and antineutrophil cytoplasmic antibody were negative in all patients apart from one with biopsy proven leucocytoclastic vasculitis who had a transiently positive antineutrophil cytoplasmic antibody. Antistreptolysin O titre was tested in only two out of the five patients and was negative.\nMean forced expiratory volume1 was 30% (range: 18–55) of the predicted value. In four patients (57%), the forced expiratory volume1 was lower at the time of presentation with rash compared to the baseline forced expiratory volume1 values for the previous two years.\nAll patients received antibiotics for a presumed pulmonary exacerbation at onset of the rash. Three patients received specific treatment for vasculitis (prednisolone ± additional immunosuppressive) after non-resolution of the rash.