Case Presentation
An eight-year-old HIV-negative boy presented with a history of chronic
cough and weight loss over a one-year period. He had no known adult
tuberculosis (TB) contacts. During this time he was seen at his local
clinic on three occasions and received a five-day course of oral
amoxicillin on two of these visits.
His mother reported that he also complained of tooth ache but decided to
forego an assessment by the dentist. On examination his body mass index
was 18.5 (normal WHO Z score) and he had clubbing of the fingers and
toes. Severe dental caries were observed. On respiratory examination,
his chest wall was normal, but there was dullness to percussion in the
right middle and lower lobe areas.
His Mantoux skin test was negative.
Antero-posterior and lateral chest X-rays (Figure 1 A and B) performed
on admission demonstrated cut-off of the bronchus intermedius with dense
consolidation of the right lower and middle lobes. Contrasted computed
tomography (CT) scans of the chest (Figure 1 C-E) performed four days
after the plain radiographs, confirmed abrupt narrowing of the bronchus
intermedius as well as dense air-space disease in the right lower and
middle lobes, with a large confluent area of central low density lacking
parenchymal structures and vessels indicative of necrosis without gas
formation. Further features included a small effusion at the right base,
and large non-necrotic, non-enhancing lymph nodes in the superior
mediastinal, right paratracheal, subcarinal and hilar areas.
Bronchoscopy was performed with a 4.9mm scope with a 2.2mm working
channel (Olympus BF-H1100 Broncho videoscope). There was complete
obstruction and narrowing of the bronchus intermedius (Figure 1 F).
Yellow, solid-appearing material was present in the bronchus intermedius
with only the right upper lobe bronchus patent. A biopsy of the material
was taken and the obstruction was relieved by meticulously removing the
adherent soft material. Once the distal part of the right lower lobe
bronchus could be reached, the presence of foreign bodies and tumours
were excluded. Due to significant bleeding, the procedure had to be
interrupted several times to allow for effective ventilation.
Following biopsy and clearing of puss and blood by suctioning through
the bronchoscope, a 5.5mm cuffed endotracheal tube (ETT) was placed in
the trachea. Despite ventilation pressures of 30-40mmH2O, there was very
little chest expansion which indicated further obstruction. A 5cm long
yellow mass clot, too thick to pass through the bronchoscope working
channel, was removed with a suction catheter after which ventilation
returned to normal.
The tissue and broncho-alveolar lavage (BAL) specimens were sent for a
full laboratory workup including cytology, histology, microbiology and
virology.
The cytology on BAL showed clumps of amorphous material in which red
filamentous organisms could be seen. Periodic acid–Schiff (PAS) and
Brown-Hopps Gram stains revealed these to be gram-positive filamentous
bacilli which morphologically suggested Actinomyces species
(Figure 1 G–L). The differential count on the BAL fluid was as follows:
macrophages: 0%, lymphocytes: 4%, neutrophils: 96%, and eosinophils:
0%.
The histology showed necrotic tissue that had a slight fibrillary
character . There was a focal area suggestive of a
Splendore-Hoeppli effect with the filaments lining up close to the
surface and interacting with inflammatory cells.
Brown-Hopps stain showed gram-positive filamentous organisms which were
Ziehl-Neelsen (ZN) negative, PAS positive and negative on Kinyoun
acid-fast stain (Figure K and L). The Gram stain showed more than 20
polymorphonuclear leucocytes per low power field, with abundant
filamentous gram-positive bacilli (Figure M).
This morphology together with the clinical information, would indicate
an organism ball and sulphur granules. The filamentous nature of these
bacteria, together with being ZN(-) and PAS(+) is highly suggestive of
actinomycosis.
Bacterial culture produced a mixed growth and, despite 14 day aerobic
and anaerobic incubation, failed to isolate Nocardia or Actinomyces . The Xpert® MTB RIF/Ultra (Cepheid,
CA, USA), TB culture and the respiratory multiplex PCR was also
negative.
The child’s immunological work-up was normal. He was treated for 14 days
with intravenous amoxicillin/clavulanic acid and then changed to oral
amoxycillin and he was referred to a dentists who removed the affected
teeth. At a 3-month follow-up visit he had significant clinical and
radiological improvement. He will be followed up regularly to decide if
surgical intervention is necessary and will be treated with oral amoxycillin therapy for
at least 12 months.