Journal of Pediatric Surgery 54 (2019) 1938–1940

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Journal of Pediatric Surgery

Surgical management of pulmonary aspergillosis in pediatric population

a r t i c l e i n f o a b s t r a c t

Article history: Introduction: Aspergillosis is an invasive infection that affects mainly immunocompromised patients, and the
Received 3 July 2018 lung is the most common site of infection. We reviewed our experience with the surgical management of pulmo-
Received in revised form 3 January 2019 nary aspergillosis (PA) in pediatric population at a tertiary care institution.
Accepted 3 January 2019 Methods: A retrospective chart review was conducted for pediatric patients (b16 years) who were managed for
PA over the period from January 2001 to December 2016. Demographic, clinical, and outcome data were
Key words:
retrieved, and descriptive data were generated.
Pulmonary aspergillosis
Fungal infection
Results: Eighty patients had microbiologically-proven PA over the period of the study, and 38 of them (48%) had a
Immunosuppression surgical intervention during their management. Among the surgical group, there were 19 males and 19 females
Surgical treatment with a mean age of 9.4 years (0.5–16). Thirty-five patients (92%) were in an immunocompromised state (10 of
Lung infection them have had bone marrow transplant), 2 patients had cystic lung disease, and 1 had scleroderma. All had
fever and respiratory symptoms at presentation and received systemic antifungal medications. All had Comput-
erized tomography (CT) scan examinations which showed features suggestive of the disease. Surgical procedures
included lung biopsy in 13 patients (34%), segmentectomy in 12 patients (32%), lobectomy in 8 patients (21%),
debridement/cavernostomy in 3 patients (8%), and 2 patients had pneumonectomy (5%). Histopathological
examination revealed invasive fungal infection in 20 patients (53%) and aspergilloma in 18 (47%). Eleven patients
(29%) died owing to a combination of fungal infection and the underlying disease. Three other patients, not
included in the surgical series, died secondary to severe pulmonary hemorrhage while waiting for surgery. All
had cavitation on CT scan near the hilum of the lung.
Conclusions: Pulmonary aspergillosis is a severe invasive infection with a significant mortality. Early surgical
intervention is recommended in patients who do not improve on antifungal medication, especially in those
with cavitating lesions, in order to improve outcome.
Type of study: Case series.
Level of evidence: IV.
© 2019 Published by Elsevier Inc.

Aspergillosis is an invasive infection that affects mainly immuno- lesions owing to poor response to medical treatment and the high risk
compromised patients, and the lung is the most common site of infec- involved. There is scarcity of data in the literature regarding surgical inter-
tion [1]. Pulmonary aspergillosis (PA) causes a spectrum of clinical vention in such a high risk pediatric population.
syndromes in the lung that ranges from hypersensitivity to invasive We reviewed our experience with surgical management of pulmo-
aspergillosis [2]. Traditionally, such severe infections occur in sick nary aspergillosis (PA) in pediatric population at King Faisal Specialist
patients who are severely immunocompromised. Increased incidence Hospital and Research Center (A tertiary care institution) over the last
of fungal infections in immunocompromised patients is related to 15 years.
prolonged immunosuppression owing to primary diseases, chemother-
apy or prolonged use of broad spectrum antibiotics [3]. Early identifica- 1. Methods
tion and diagnosis are important to achieve a better outcome. Typically,
treatment starts with medical agents including potent systemic antifun- A retrospective chart review was conducted for pediatric patients
gal medications, while treatment duration is tailored according to (b 16 years) who were managed for PA over the period from January
patient's response and his/her immunological status [4]. Most of the 2001 to December 2016. Patients with PA, proven with a positive
latter strategies are extrapolated from the adult experience. Surgical culture (respiratory or tissue), were included. Traditionally, such
management of pulmonary aspergillosis is usually reserved to patients patients were started on antifungal medications. Patients who improve
who do not respond to medical treatment especially those with cavitating clinically (resolution of their symptoms and signs) and radiologically
(improvement of findings on imaging studies) are to continue the
⁎ Corresponding author. course of the antifungal treatment. Patients who fail to respond to anti-
E-mail address: [email protected] (S. AlShanafey). fungal treatment clinically (persistence or worsening of symptoms and

https://doi.org/10.1016/j.jpedsurg.2019.01.003
0022-3468/© 2019 Published by Elsevier Inc.
 S. AlShanafey et al. / Journal of Pediatric Surgery 54 (2019) 1938–1940 1939

Table 1 Hemoptysis and cavitation near the hilum on CT scan were considered
Characteristics of surgical and medical groups. urgent indications for surgery later in the series owing to mortalities
Characteristics Surgical group Medical group P-value encountered in patients with such presentation/findings (see below).
N = 38 N = 42 Surgical referral was for diagnostic purposes (lung biopsy) and definitive
Gender (male: female) 19: 19 18: 24 0.52 treatment (resections and debridement). Histopathological examinations
Age (years) 9.4 8.8 0.49 revealed invasive fungal infection in 12 patients (53%) and aspergilloma
Immunocompromised (N, %) 35 (92%) 38 (90%) 0.39 in 18 (47%). Two had prolonged air leak (more than 1 week). Both had
Bone marrow transplant (N, %) 10 (29%) 8 (19%) 0.22
cavitating lesions and were managed with debridement/cavernostomy.
Mortality (N, %) 11 (29%) 4 (9%) 0.01⁎
Air-leak was managed conservatively and eventually sealed in 9 and
⁎ Statistically significant.
13 days respectively postoperatively. There were 11 (30%) postoperative
deaths among the surgical group (8 had lung biopsy for consolidation,
signs) and radiologically (persistence/progression of disease on imaging 2 had resections for central cavitating lesions, and 1 had resection for
studies) are referred for surgical evaluation/intervention. Later in peripheral cavitating lesion). Three of them have had BMT and cause of
the series, patients with centrally-located cavitating lung lesions were death was a combination of fungal infection and the underlying disease
referred early for surgical evaluation/intervention to avoid severe hem- with a mean duration of 17 days postoperatively (7 ALL, 2 CGD, 1 anaplas-
orrhage and death. Demographics, presenting symptoms, radiological tic anemia, and 1 metachromatic leukodystrophy). In comparison, among
findings, type of management (especially surgical intervention), out- the medical group (42 patients), 4 died (2 ALL, 1 AML, 1 CGD), and 1 of
come, and histopathology were retrieved, and descriptive data were them have had BMT. All deaths were primarily because of their underly-
generated. Those who were treated surgically were compared to those ing disease. None of the patients died secondary to the surgical procedure
who were treated medically only. The two groups were compared or its complications. Three other patients (2 with acute lymphocytic
using t-test for continuous variables and chi-square test for proportions. leukemia and 1 with chronic granulomatous disease), not included in
The study was evaluated and approved by our institutional research the series (respiratory cultures were negative and no autopsy was
board (IRB) after been reviewed by the clinical and ethical research performed), died secondary to severe pulmonary hemorrhage while
committees (RAC # 2131021). waiting for surgery (less than 48 h). All were booked semielectively at
the 1st available operative room slots while inpatients. All presented
2. Results with mild hemoptysis and had cavitating lesions on CT scan highly sug-
gestive of PA. All were stable clinically while inpatients and had sudden
Eighty patients had microbiologically-proven PA over that period bleed that was difficult to manage. Among surgical patients who died
and 38 of them (48%) had a surgical intervention. Diagnostic specimens (11 postoperative, 3 prior to planned surgery), CT examinations revealed
included sputum (7 patients), nasopharyngeal aspirate (18 patients), central cavitating lesions in 7 (50%), consolidation in 6 (43%), and lung
bronchial lavage (33 patients), and tissue (38 patients). Some patients nodule in 1 (7%).
in the surgical group had more than one diagnostic specimen. In the
surgical group, all patients have been started on antifungal therapy 3. Discussion
with a mean of 6.7 weeks (SD 2.2) prior to the surgical intervention.
Among the surgical group (38 patients), 2 patients had cystic lung Immunocompromised patients are more prone to fungal infections,
disease, and 1 had scleroderma. The remaining 35 patients (92%) were especially aspergillosis. It seems that the invasiveness of the disease
in an immunocompromised state. The latter included the following correlates with the degree of the immunity defect. Recent reports
diagnoses: 15 acute lymphocytic leukemia (ALL), 8 acute myeloid leu- have suggested an increased incidence of invasive aspergillosis (IA)
kemia (AML), 7 chronic granulomatous disease (CGD), 4 anaplastic ane- in the recent years [5,6] with more pronounced increase in acute
mia, and 1 metachromatic leukodystrophy, and 10 (29%) of them have myeloid leukemia (AML). AML patients have a 25% chance of develop-
had bone marrow transplant (BMT) at the time of diagnosis of PA. ing IA regardless of having bone marrow transplant or not [5]. The
Among the medical group (42 patients), 2 patients had chronic lung dis- latter finding may suggest that incidence of IA increases with more
ease and 2 patients had cystic lung disease. The remaining 38 patients immunocompromisation owing to aggressive chemotherapy protocols
were in an immunocompromised state (ALL 23, AML 7, CGD 3, anaplas- developed over the recent years. On the other hand, PA has also been
tic anemia 3, and 2 patients had rhabdomyosarcoma on chemotherapy). reported rarely in immunocompetent patients. Scleroderma patients
A comparison between the surgical and medical group is shown in are prone to pulmonary fibrosis and rarely reported with PA [7,8].
Table 1. All had fever and respiratory symptoms at presentation and Other cystic and chronic lung diseases were also associated with
2 presented with mild hemoptysis. All were started on systemic antifun- PA [8–10]. The chronicity of the latter diseases and the frequent use of
gal medications before surgery. Computerized tomography (CT) scan antibiotics may have made them more prone to such invasive fungal
was performed for all patients. In the medical group, CT scan showed disease.
mainly diffuse pneumonitis and consolidation. While in the surgical Apart from diagnostic lung biopsies, surgical management of this in-
group, 24 (67%) had lung consolidation, 23 (64%) had lung cavitation, vasive disease is usually reserved for severe forms that do not respond
and 3 (8%) had lung nodules. well to medical treatment and certain cases with a cavitation process
Surgical procedures performed are shown in Table 2. Indications for [11]. Specifically patients who have large cavities in proximity to
surgery included failure of medical treatment defined as persistence or blood vessels (proximal to the hilum) are at high risk for bleeding and
worsening of symptoms and progression of the disease radiologically. death [12]. For the latter, other means of management other than
surgery were not successful. Systemic antifungal agents were not effec-
tive for the cavitating form of the disease owing to poor penetration
Table 2 [11]. Moreover, angiography is not highly successful in identifying
Surgical procedures performed (38 patients).
bleeding vessels in patients presenting with hemoptysis [12]. Attempts
Surgical procedure Frequency Mortality at embolization to control bleeding in such cases were not usually suc-
Lung biopsy 13 (34%) 8 (61%) cessful and recurrence rate was high owing to the existence of collateral
Segmentectomy 12 (32%) 1 (8%) circulation. Many surgical procedures are performed for patients with IA
Lobectomy 8 (21%) 2 (25%) refractory to medical treatment. These include segmentectomy, lobec-
Debridement/cavernostomy 3 (8%) 0 tomy, pneumonectomy, cavernostomy and debridement depending
Pneumonectomy 2 (5%) 0
on the extent of disease and the status of the patient [13,14]. In our
1940 S. AlShanafey et al. / Journal of Pediatric Surgery 54 (2019) 1938–1940

experience and others, segmentectomy and lobectomy by far were An early surgical intervention is recommended in patients who do not
the most preferable effective techniques regarding complications and respond to medical treatment or those who harbor cavitating lesions.
outcome [14].
Published series in adult patients have reported that the most
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