Case presentation
In August 2018, a 32-year-old pregnant mother at her 34 weeks of gestation was referred to Dr. Shariati Hospital in Mashhad, Iran, due to weight loss, fever, persistent, coughing and dyspnea. She was diagnosed by sputum smear and culture examination to have pulmonary tuberculosis. The patient was treated for category I tuberculosis, and her symptoms became gradually negative in accordance with the sputum smear results. However, she delivered about one month later at 41 weeks gestation. Despite receiving prophylactic TB therapy (isoniazid), 55 days after his birth, the child (boy infant) was affected to fever (38.3°C), anorexia and a painful swollen lesion in the neck area, and was taken to Dr. Shariati Hospital in Mashhad. His parents had no consent for lumbar puncture. Therefore, since the mother had tuberculosis during pregnancy, the baby was also diagnosed as a “TB Case” and treated with isoniazid and rifampicin. Although the neck mass biopsy results showed evidence of inflammation but they were negative for the acid-fast bacilli. The child’s cervical swelling did not improve after about 4 months of anti-tuberculosis therapy, and had a pale colored discharge.
The purified protein derivative (PPD) test indicated about 3 mm, when he was 7 months old. Moreover, the patient was also negative for the HIV, HBV and HCV tests. The patient had no evidence of organomegaly. CBC results included WBC: 24,000 / mm3, RBC: 5200 / μm, Hb: 11.5 g.dl, HCT: 37.4% and Platelets: 535,000 / mm3. The serum levels of IgG, IgM and IgA were 1500, 122 and 104 mg.dl, respectively. The parents of his patient were not consent for lumbar puncture of baby and disseminated TB was ignored based on the radiological findings and negative culture of blood forMtb . The sonography analyses of the internal organs such as liver, bile duct, pancreas, kidney and bladder were normal and no evidence of pleural effusion; Chest X-ray (CXR) images of the patient’s lung showed normal results (Figure 1).
The patient underwent FNA (Fine Needle Aspiration) and the patient’s biopsy specimen was sent to Tuberculosis Reference Laboratory in Mashhad for cytopathology and culture studies. Based on reported results from the neck mass biopsy, numerous evidences of multiple necrotizing granuloma with caseous lesion were observed; Ziehl-Neelsen staining results confirmed the existence of acid-fast bacilli. The results of the “QuantiFERON assay” also identified the presence of an immunological response to Mtb infection.
The standard proportional method analysis was carried out according to the Clinical and Laboratory Standards Institute on Lowenstein-Jensen medium and resistance was observed to isoniazid, rifampicina and ethambutol. According to the GeneXpert MTB-RIF assay, the Mtb isolate was resistant to rifampicin. In addition, based on the drug molecular susceptibility test (DST) results performed by DNA sequencing, the considered isolate was resistant to rifampicin, isoniazid and ethambutol and was introduced as an MDR-TB case. Due to the importance of identifying source of infection, the Mtb isolates of the studied infant and her mother underwent genetic fingerprinting subjected to IS6110-RFLP and Spoligotyping. It was found that the infant isolate was from the Beijing lineage, whereas that of her mother’s was from the mixed infection with Beijing and Delhi/CAS lineages. Confirmation of IS6110-RFLP results by Spoligotyping suggested that the mother had multiple strains as the consequence of recent transmission. It has been more probable that the patient was infected with Mtb as congenitally route. However, the patient was treated with a combination therapy including surgical drainage and antibiotic therapy by ethionamide, moxifloxacin, amikacin and linezolid. Other members of the patient’s family also received prophylactic MDR-TB (with moxifloxacin). After one year and regular follow-up of the patient, it was found that the patient had fully recovered and no signs of reactivation were observed in the patient, his mother, and their other family members.