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.