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The continuity of patient care post-hospital discharge is a
This clinical profiling is important as the transition of
Renal failure
accident

Cerebrovascular
Infectious
Respiratory
accident
Cardiac
Other
mellitus
Gastrointestinal
Renal failure
Diabetes
Cerebrovascular

Taken together, these data demonstrate that T2DM patient
0.003
19 (55.8%)
4 (11.7%)
0.043
7 (16.2%)
25 (58.1%)
0.05
13 (17.1% )
47 (61.8%)
0.061
3.88 ± 1.53
3.72 ± 1.73
7.45 ± 6.07
0.27
8.36 ± 5.4
n=106 (69.3%)
n=25 (16.3%)
no adherence
p value
full adherence
Original articles Original articles mellitus Diabetes 11(32.3%) 11 (25.5%) 16 (21.0%) 4.68 ± 1.81 9.82 ± 8.45 n=22 (14.4%) partial adherence Gastrointestinal Other Compliance Respiratory Cause of hospitalization Yes (n=34) Yes (n=43) Yes (n=76) 3.88 ± 1.73 7.94 ± 6.38 n=153 Total Infectious Cardiac 0% 20% 40% 60% 80% 0% Background AF Background CHF Background CHD background diseases Number of hospitalization Length of B A better
#
no differences in short-term complication rates, anastomotic eight of whom presented with a single recurrence. One patient says are limited to the panel of antigens incorporated in the test between IIF and ESC for SLE, Sjögren’s syndrome, SSc, MCTD, and
leak, or anastomotic stricture were found between the thora- (1.3%) died at 17 years of age due to respiratory insufficiency system. Therefore, the two techniques show differences in the PM/dermatomyositis was 93% vs. 98%, 75% vs. 81%, 100% vs. 50%,
coscopic and open approaches [14]. and sepsis after three episodes of recurrent TEF. detection of ANA. 100% vs. 100%, and 100% vs. 100%, respectively. Furthermore, IIF
To the best of our knowledge, later factors that may influ- Demographic data, spirometry, and CT findings of the In our study, we observed ECS positive /IIF negative results in 2.2% showed a positive result in all patients with an ANA-associated dis-
ence recurrence of TEF have not been studied. However, we patients are presented in Table 1. Comparison of the groups of and ECS negative /IIF positive results in 16.9% of 1708 subjects. The order in remission, whereas ESC was positive in only 22% [10]. In a
have encountered several patients in whom recurrence of TEF patients with and with no recurrence of TEF is presented in Table ECS positive /IIF negative specimens (except from dsDNA-, Ro/SSA-, trial comparing two solid-phase assays with IIF, the area under the
was diagnosed concurrently or shortly after viral bronchiolitis. 2. As can be seen, the groups were similar in terms of age, gender, and Jo-1 antibodies) mostly contained low antibody con- receiver operating characteristics curve (AUC) for Sjögren’s syn-
Such an association has not been reported. VACTERL association, and spirometry. Moreover, the anatomic centrations of histone, SSB/La, Sm, Scl-70, and U1-RNP. In drome was higher for the solid-phase assays than for IIF; whereas
The objective of this study was to describe the incidence and type of TEF, length of the atretic gap, type of surgery performed, the ECS negative /IIF positive group, Combined screening with for SSc, the AUC was higher for
the risk factors of recurrent TEF in a tertiary pediatric hospi- except from one centromere-B immunofluorescence on Hep-2 cells and IIF than for a solid-phase test [15].
tal. In addition, we aimed to assess the possible association of Table 1. Patients characteristics antibody in a patient with limited Finally, in a recently published paper
Elia connective tissue disease screen
recurrent TEF and bronchiolitis. patient characteristics scleroderma, only antibodies not assay enhances the diagnostic accuracy by Willems and co-authors [8], IIF
Mean age, years (median; range) 8.2 ± 5.67 (8; 0.5–28) included in the ECS panel (e.g., for antinuclear antibodies screening and ESC were positive in 90.4% and
69.9% of SLE, 100% and 84.1% of
histone, nucleosome, Pl-12 and
PATIENTS AND METHODS Gender, male 41 (55%) AMA-M2) were detected [9]. In a large Italian study population, SSc, 86.7% and 93.3% of Sjögren’s syndrome, 88.2% and 52.9% of
A retrospective review was conducted of patients who under- Concurrent anomalies 29 (39%) ECS positive /IIF negative results occurred in 2% (mainly Ro52, Ro60, PM/dermatomyositis, and in all cases of MCTD.
VACTERL
went a previous surgery for TEF and who were followed in the CHARGE 1 (1.3%) dsDNA, PM/Scl, and Jo-1), and ECS negative /IIF positive results were
pediatric pulmonary institute of our hospital between January Feingold syndrome 1 (1.3%) found in 31% [11]. These findings are in accordance with previ- wHAT SHOulD BE THE pREfERRED SCREEnIng STRATEgy fOR AnA?
1 (1.3%)
Concurrent CCAM
2007 and December 2016. The institutional board reviewed and Recurrence of fistula 9 (12%) ously published studies that revealed that ECS is able to detect Since solid-phase assays and IIF differ significantly in the
approved the study. Patients were excluded if the information Died antibodies that are missed by IIF, and vice versa, IIF may detect sensitivity and specificity for various ANA-associated autoim-
in the hospital medical record was insufficient. 1 (1.3%) relevant antibodies that are missed by ECS [5,8,12,13]. mune disorders [15], it seems likely to establish a combined
Perioperative data obtained included demographic data, FEV1 (% predicted) n=15 68 ± 20.7 There is another point that merits mention. It has been shown screening algorithm incorporating both techniques, either in a
Mean ± SD
TEF as a solitary finding or as part of an association, open or Median (range) 74 (30–96) that even antibodies that are included in the panel of automated sequential or a parallel screening approach. Bossuyt and Fieuws
thoracoscopic repair, need for prolonged respiratory assistance Computed tomography (n=20) assays may be missed. For example, Parker and colleagues [7] [14] recommended that favorable strategies should be disease-
post-surgery, and length of the atretic gap. Postoperative data Normal lung fields 6 6 observed that the ECS is a sensitive method for the detection of dependent. In their study, the best diagnostic strategy for SLE
Bilateral bronchiectasis
included recurrence of the TEF, occurrence of gastroesopha- Uneven ventilation and atelectasis 4 anti-PM-Scl, anti-Mi-2, anti-PCNA, and anti-Rib-P antibodies, and Sjögren’s syndrome was to perform both tests on all samples;
geal symptoms, number of hospitalizations due to respiratory Mediastinal collection 1 1 but is a suboptimal screening tool for anti-fibrillarin and anti- whereas for SSc, screening with IIF and performing a solid-phase
Post lobar resection
reasons, number of episodes of viral bronchiolitis, and positive Bilateral infiltrates 2 RNA polymerase III antibodies, with a detection rate of 68% assay on IIF-positive samples was comparable to both IIF and 131118-COHANIM - 131118-COHANIM | 4 - B | 18-11-13 | 11:24:13 | SR:-- | Magenta
polymerase chain reaction (PCR) for respiratory viruses. CCAM = congenital cystic adenomatoid malformation, CHARGE = coloboma, and 67%, respectively. solid-phase assay on all samples. Bizzaro et al. [11] showed that 131118-COHANIM - 131118-COHANIM | 4 - B | 18-11-13 | 11:24:13 | SR:-- | Yellow #131118-COHANIM - 131118-COHANIM | 4 - B | 18-11-13 | 11:24:13 | SR:-- | Black 131118-COHANIM - 131118-COHANIM | 4 - B | 18-11-13 | 11:24:13 | SR:-- | Cyan
We used the term “recurrence of TEF” to refer to all patients heart defects, choanal atresia, retarted growth, genital abnormalities, the association of solid-phase screen assays to IIF increases the
ear abnormalities, FEV1 = forced expiratory volume in 1 second, SD =
who had a fistula after a prior operation. This term includes standard deviation, VACTERL = vertebral defects, anal atresia, cardiac defects, DIffEREnCES BETwEEn ECS AnD IIf In THE SEnSITIVITy fOR sensitivity from 89.2% to 97.4% and the specificity from 64.6%
tracheoesophageal fistula, renal anomalies, and limb abnormalities
recurrence of TEF in the same location as the original fistula, AnA-ASSOCIATED SySTEMIC AuTOIMMunE DISORDERS to 98.4% in serological tests for ANA screening. In addition,
fistula de novo in a different location, or a second TEF that may Based on the different diagnostic sensitivity for various ANA an analysis of costs demonstrated that the combination of the
have been missed prior to the first operation. Table 2. Statistical comparison of patients with and without subtypes, several studies reported that ESC and IIF also show a two techniques represents a cost-effective diagnostic pathway,
Spirometry data and main computed tomography (CT) recurrence of tracheoesophageal fistula different diagnostic performance for ANA-associated autoim- reducing the global costs for the immunoserological diagnosis of
findings were recorded when available. No recurrence of Recurrence P mune disorders. ANA-associated autoimmune disorders by 22% [11].
TEF (n=65) of TEF (n=9) value In one study, the sensitivity was higher for IIF than ESC for
Age, years (range) 7 (4–11) 8 (1.5–11.5) 0.97
STATISTICAl METHODS SLE and SSc, but not for Sjögren’s syndrome. A higher specificity COnCluSIOnS
Statistical analyses were performed using IBM Statistical Package Gender, male 36 (55%) 5 (56%) 1.00 was observed for the ESC [14]. Several studies have confirmed that ESC and IIF differ in the
for the Social Sciences statistics software, version 21 (SPSS, IBM Patients hospitalized with 32 (49%) 7 (77%) 0.16 In a study conducted by our group, ECS had a 100% sensitiv- diagnostic sensitivity for the various types of ANA-associated
respiratory symptoms
Corp, Armonk, NY, USA). Descriptive statistics were used for the VACTERL association ity for Sjögren’s syndrome, SSc, and MCTD. The sensitivity for systemic autoimmune diseases. Combined screening with IIF
demographic variables, clinical parameters, spirometry, and CT 27 (42%) 2 (22%) 0.46 Sjögren’s syndrome was higher for Antinuclear antibodies (ANA) screening and ECS enhances the diagnostic
findings. Differences between the groups with and those without FEV1 % predicted, mean ± SD 69.7 ± 22.6 (n=14) 62 ± 17 (n=3) 0.51 ESC than for IIF (94%). IIF had solely by Elia connective tissue disease sensitivity and specificity for ANA,
recurrent TEF in the quantitative parameters were measured by Surgery 42 (65%) 5 (56%) 0.72 a higher diagnostic sensitivity for screening assay might be justifiable in and beyond that, markedly reduces
Open
Mann–Whitney U tests and Fisher’s exact tests for categorical Thoracoscopy 23 (35%) 4 (44%) 0.72 SLE, indetermined connective tis- the costs for the laboratory diagno-
parameters. P < 0.05 was considered as statistically significant. Prolonged respiratory assistance 18/61 (30%) 1/7 (14%) 0.66 sue disease, Raynaud’s syndrome, a setting with a low pretest probability sis for ANA-associated disorders.
Gastrointestinal symptoms 40 (62%) 6 (67%) 1.00 and limited scleroderma, compared for ANA-associated disorders Screening for ANA solely by ECS
RESULTS Anatomic abnormality (C type) 60 (92%) 9 (100%) 1.00 to the ESC (100% vs. 80%; 100% vs. 75%; 89% vs 57%; 100% vs. might be justifiable in a clinical setting with a low pretest prob-
88.9%, respectively) [9].
ability for ANA-associated systemic autoimmune disorders.
Atretic gap, cm (range) 1.56 (0–4) n=42 1.6 (1–3) n=5 1.00
Seventy-seven post-TEF repair patients were identified. Three The authors of a recently published study reported an over- However, in cases of a clinical suspicion of ANA-associated
patients were excluded due to insufficient data in their medical FEV1 = forced expiratory volume in 1 second, TEF = tracheoesophageal all positivity rate for ANA-associated autoimmune disorders of disease and a negative ECS, additional IIF should be performed PERFECTOR
fistula, VACTERL= vertebral defects, anal atresia, cardiac defects,
records. Nine patients (12%) experienced a recurrence of TEF, tracheoesophageal fistula, renal anomalies, and limb abnormalities 90% for IIF and 92% for the ESC. In that study, the positivity rate [1,8,10].
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