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\begin{document}

\title{Real-time ultrasound guided segmental bronchoscopic insufflation in a
Tay-Sachs patient with atelectasis}



\author[1]{Matej Šapina}%
\author[1]{Bojana Olujić}%
\author[1]{Tihana Nađ}%
\author[1]{Hrvoje Vinković}%
\author[1]{Zdravka Krivdić Dupan}%
\author[1]{Nikolina Hamidović}%
\author[1]{Mia Damašek}%
\author[1]{Krešimir Milas}%
\author[1]{Željko Zubčić}%
\author[1]{Ivan Erić}%
\affil[1]{Klinicki bolnicki centar Osijek}%


\vspace{-1em}



  
  \date{May 25, 2024}


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Real-time ultrasound guided segmental bronchoscopic insufflation in a
Tay-Sachs patient with atelectasis

Matej S\selectlanguage{english}apina, MD, PhD\textsuperscript{1,2,3*}, Bojana Olujic,
\selectlanguage{english}MD\textsuperscript{1,2}, Tihana Na\selectlanguage{polish}đ, \selectlanguage{english}MD\textsuperscript{1,2}, Hrvoje
Vinkovic, \selectlanguage{english}MD\textsuperscript{1,2}, Zdravka Krivdic \selectlanguage{english}Dupan,
MD\textsuperscript{1,2}, Nikolina Hamidovi\selectlanguage{polish}ć, \selectlanguage{english}MD\textsuperscript{1}, Mia
Dama\selectlanguage{polish}š\selectlanguage{english}ek, MD\textsuperscript{1,2}, Kres\selectlanguage{english}imir Milas, MD\textsuperscript{1},
Z\selectlanguage{english}eljko Zubc\selectlanguage{english}ic, \selectlanguage{english}MD, PhD\textsuperscript{1,2}, and Ivan Eric, \selectlanguage{english}MD,
PhD\textsuperscript{1,2}

\textsuperscript{1}University hospital center Osijek, 31000 Osijek,
Croatia

\textsuperscript{2}Medical faculty Osijek, 31000 Osijek, Croatia

\textsuperscript{3}Faculty of dental medicine and health Osijek, 31000
Osijek, Croatia

\textsuperscript{*}Corresponding author: sapina.matej@yahoo.com

Phone: +385 31 512 279

Keywords: bronchoscopy, atelectasis, Tay-Sachs disease

Introduction

To the Editor,

Atelectasis is the collapse of lung tissue and is a common problem in
patients suffering from severe neurological diseases. Treatment depends
on the underlying cause, and ranges from respiratory physiotherapy,
mucus clearance methods, and in some cases bronchoscopic interventions.
If left untreated recurrent infections may occur, along with impaired
gas exchange and in the long run fibrosis of the affected
area\textsuperscript{1}.

A female patient at the age of 3 years with Tay-Sachs disease and
consequent severe global psychomotor delay and epilepsy was initially
admitted due to a respiratory infection to the neuropediatric
department, where she developed an epileptic status and desaturation,
prompting a transfer to the Intensive Care Unit. The refractory
epileptic status resolved only with several antiepileptics, which lead
to intubation requiring invasive mechanical ventilation, after which the
patient stabilized.

An attempt to reduce sedation the next day resulted in repeated
seizures. On the third day, inflammatory parameters increased, with a
radiological finding suggestive of pneumonia and a large left
atelectasis confirmed by ultrasound. Antibiotics were empirically
started, and Moraxella catarrhalis and Adenovirus in the respiratory
tract were obtained in the cultures.

Lung ultrasound showed a large consolidation in the dorsal upper two
thirds of the left hemithorax. Due to the size of the atelectasis, we
decided to perform bedside bronchoscopy.

A single use paediatric flexible bronchoscope (Ambu\selectlanguage{ngerman}® aScope\selectlanguage{english} 5 Broncho)
was introduced through the ETT. The tracheobronchial morphology did not
show any obstructions. We applied our recently published modified
segmental insufflation technique, however without the use of
surfactant\textsuperscript{2}.

First, a bronchoalveolar lavage was performed for microbiological and
cytological analysis, followed by ultrasound guided bronchoscopic
treatment of the affected area. The patient was positioned on the right
lateral decubitus position. As described previously, a cuff pressure
manometer (AG Cuffill Cuff) and a 50 ml syringe were attached to a
three-way stopcock connected to the bronchoscope's working channel.
Pressure controlled segmental insufflation of the lingula and upper lobe
was performed directly observing an aeration of the atelectatic areas on
the ultrasound with no complications.

Subsequent ultrasound monitoring resulted in total expansion of the
affected lung and resolution of inflammation, confirmed radiologically
(Figure 1.). The patient could be safely extubated and transferred to
the wards. No reoccurrence of atelectasis was observed.

Conclusion

The treatment of pediatric atelectasis poses a challenge, with limited
approaches available and a lack of evidence-based guidelines to guide
clinical decision-making. A question that remains to be answered is not
only the mode of treatment, but timing. Even with bronchoscopic
treatment, different methods exist. In obstructive atelectasis, the
approach is simple - to eliminate the obstruction, however,
nonobstructive atelectasis remain challenging. Bronchoscopic lung
insufflation technique has been explored in adults, with limited cases
reported in children\textsuperscript{3}. Several case reports utilized
pressure-controlled insufflation with or without surfactant in long
standing atelectasis as an add on treatment\textsuperscript{2; 4}.

We believe that patients with severe neurological diseases require
special attention regarding their susceptibility for developing
atelectasis\textsuperscript{5}. Our approach is early bronchoscopic
pressure-controlled insufflation in non-obstructive atelectasis.
Depending on the duration, we additionally instill surfactant in long
standing ones.

Ultrasound is recently being used in lung recruitment maneuvers to
monitor lung expansion, however, it was only recently reported to be
utilized for segmental insufflation monitoring in a spinal muscular
atrophy type I patient\textsuperscript{4}. However, to our best
knowledge, our case report is the first successful use on a Tay-Sachs
patient using real time ultrasound monitoring during pressure-controlled
segmental bronchoscopic insufflation. The benefits of ultrasound
guidance is the direct visualisation of lung expansion reducing the need
for fluoroscopic radiation exposure of visualisation and further
monitoring. Besides it increases the safety of the procedure allowing
for detection of development of pneumothorax.

In conclusion, pressure controlled real-time ultrasound guided
bronchoscopic segmental insufflation, with its low-cost, lightweight,
and flexible setup, seems to be a safe and promising new modality for
treatment of non-obstructive atelectasis, especially in children
requiring complex care.

FIGURES

Figure 1. On the left a large left sided atelectasis can be seen causing
the mediastinal shift. On the right complete regression of the
atelectasis is observed before extubating

References

1. Ogake S, Bellinger C. 2020. Role of bronchoscopy in atelectasis.
Clinical Pulmonary Medicine. 27(1):30-32.

2. S\selectlanguage{english}apina M, Olujic B, Na\selectlanguage{polish}đ \selectlanguage{english}T, Vinkovic H, Dupan ZK, Bartulovic I, Milas
K, Kos M, Divkovic D, Zubc\selectlanguage{english}ic \selectlanguage{english}Z. 2023. Bronchoscopic treatment of
pediatric atelectasis: A modified segmental insufflation-surfactant
instillation technique. Pediatric Pulmonology.

3. Abu-Hasan MN, Chesrown SE, Jantz MA. 2013. Successful use of
bronchoscopic lung insufflation to treat left lung atelectasis.
Pediatric Pulmonology. 48(3):306-309.

4. Esteban IA, Osona B, Ballest\selectlanguage{ngerman}ín AS, Rubio CC, de Mendiola JMFP, Gómez
VA, López ME, Vicente JCDC. 2024. Bronchoscopic segmental alveolar
recruitment in a patient with spinal muscular atrophy and massive
atelectasis. Pediatric pulmonology.

5. St\selectlanguage{english}-Laurent A, Zysman\selectlanguage{english}-Colman Z, Zielinski D. 2022. Respiratory
prehabilitation in pediatric anesthesia in children with muscular and
neurologic disease. Pediatric Anesthesia. 32(2):228-236.\selectlanguage{english}
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