Clinical features of Kawasaki disease with atlantoaxial rotatory fixation

doi:10.21203/rs.3.rs-1441197/v1

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Clinical features of Kawasaki disease with atlantoaxial rotatory fixation

https://doi.org/10.21203/rs.3.rs-1441197/v1

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Purpose

Atlantoaxial rotatory fixation (AARF) is a rare complication of acute Kawasaki disease (KD). Early diagnosis and intervention are important for AARF because delayed diagnoses may incur neurological sequelae. However, previous studies on AARF associated with KD are limited, and its clinical characteristics and course are unknown. This study aimed to examine the clinical features and treatment course of KD with AARF using a Japanese national inpatient database.

Methods

Using the Diagnosis Procedure Combination database, we identified KD patients who received Intravenous immunoglobulin (IVIG) treatment between July 2010 and March 2020. The clinical characteristics of KD patients with AARF and their risk factors were evaluated using multivariable logistic regression analysis. We also examined the relationship between AARF, the proportion of coronary artery abnormalities (CAAs), IVIG resistance, length of stay, and medical costs.

Results

We identified 71,913 patients with KD, 166 of whom had AARF. The AARF group had older age, heavier bodyweight, and atypical KD. In multivariable analysis, AARF was associated with older age (odds ratio [OR], 1.24; 95% confidence interval (CI), 1.19–1.29), lower body mass index (OR, 0.89; 95% CI, 0.82–0.96), and atypical KD (OR, 1.95; 95%CI, 1.12–3.40). AARF was not associated with CAAs (OR, 0.73; 95%CI, 0.23–2.32) and IVIG resistance (OR, 1.05; 95%CI, 0.74–1.49). However, AARF was associated with higher medical costs (difference, US$1064; 95%CI, 346–1781) and longer hospital stay (difference, 3.1 days; 95%CI 1.7–4.4).

Conclusion

AARF in patients with acute KD should be considered if cervical symptoms present in older patients with atypical KD.

atlantoaxial rotatory fixation

coronary artery abnormalities

intravenous immunoglobulin

Kawasaki disease

Atlantoaxial rotatory fixation (AARF) is a rare complication of acute Kawasaki disease and early diagnosis, and intervention are important because delayed diagnoses may incur neurological sequelae. Few studies have shown its clinical characteristics and course.

The patients with AARF were associated with older age, lower body mass index, and atypical Kawasaki disease. AARF was not associated with coronary abnormalities. However, AARF was associated with higher medical costs and longer hospital stay.

Kawasaki disease (KD) is an acute systemic vasculitis that causes acquired heart disease in children in developed countries [1]. Atlantoaxial rotatory fixation (AARF) is a condition with a fixed ipsilateral tilt and rotation of the head to the contralateral side, resulting in torticollis that is called the “cock-robin sign” [2]. Non-traumatic AARF in children has been implicated in all cervical inflammation and surgeries, and conservative treatment is the most successful with rapid diagnosis [3]. However, if patients remain undiagnosed for a long time, the atlantoaxial joint may result in severe deformity, which may require complex surgical treatment [4–6]. Therefore, the early diagnosis and treatment of AARF are important, and delayed diagnosis leads to neurological sequelae, such as radiculopathy, paralysis, and death [7]. Previous studies reported that torticollis occurred as a rare complication of KD affecting older female children [8–12], and conservative treatment showed satisfactory outcomes in KD patients with AARF without any surgical treatment [12]. However, their sample sizes were small, and the clinical features and course of AARF remain unknown. There is also a lack of knowledge about the clinical features that make KD patients more likely to have AARF and whether patients with KD complicated by AARF have poorer short-term outcomes. Using a national inpatient database in Japan, this study aimed to (i) compare the clinical features of KD patients with and without AARF, (ii) examine factors affecting the occurrence of AARF in KD patients, and (iii) determine whether KD patients with AARF have a worse short-term prognosis than those without AARF. In addition, KD patients with AARF were examined in detail to determine whether orthopedic treatment was provided during hospitalization.

Data source

This retrospective cohort study used the Diagnosis Procedure Combination database [13]. All 82 academic hospitals were required to participate in the database. Data for approximately 8 million hospitalized patients of all ages are collected every year, equivalent to approximately 50% of the total number of acute-care hospitalizations in Japan. The database included the following information: unique identifiers of hospitals, patient baseline characteristics, diagnosis at admission, comorbidities at admission, and complications after admission recorded with text data in Japanese and International Classification of Diseases, Tenth Revision (ICD-10) codes. The database also contains information on medical procedures and treatments (including drug administration, device use, and surgical and nonsurgical procedures) based on the original Japanese codes, length of stay, discharge status, and medical costs during hospitalization. Attending physicians must confirm all diagnoses and comorbidities.

Furthermore, because accurate reporting is linked with the payment system in Japan, attending physicians and hospitals are required to accurately report diagnoses and comorbidities. This study was approved by the Institutional Review Board of the University of Tokyo (approval number: 3501-(3); December 25, 2017). The requirement for informed consent was waived because of the anonymity of the data.

Participants

We used this database to identify patients diagnosed with KD (ICD-10 code: M303) between July 2010 and March 2020. We included patients who received at least 2 g of intravenous immunoglobulin (IVIG). Atypical KD is generally defined as KD that does not meet the full criteria. Typically, patients with atypical KD have fever (even within 5 days) and only two or three major clinical manifestations. In the database, diagnoses were recorded using Japanese text and ICD-10 codes, and physicians described whether KD was the atypical type in the Japanese text. We excluded those with a weight < 3 kg, missing data, or "suspected" KD.

We identified AARF patients with a diagnosis of AARF (ICD-10 code: M4361) or the text data of "AARF" in Japanese. KD patients who were not diagnosed with AARF but were treated conservatively for AARF were also regarded as AARF patients.

Statistical analysis

We compared patient backgrounds between patients who had AARF and those who did not.

The patients’ backgrounds were investigated, including age, sex, weight, height, body mass index (BMI), hospital days at initial IVIG (i.e., the interval from the admission day to the day when the initial IVIG was administered), atypical KD or not [14], type of hospital (academic hospital or not), complex chronic conditions (classified based on the definitions reviewed in a previous study [15]), transportation by ambulance, Japan Coma Scale at admission, transportation from other hospitals, and hospital volume. The Japan Coma Scale score at admission was categorized into two groups: alert and not alert. The Japan Coma Scale assessment was previously shown to be associated with the Glasgow Coma Scale assessment [16]. The hospital volume was defined as the number of patients with KD at each hospital every year. We separated the included patients into tertiles of hospital volume so that the number of patients in each group was almost equal.

Categorical variables are shown as numbers and percentages and were compared between groups with and without AARF using Fisher’s exact test. Continuous variables are shown as the mean and standard deviation (SD) or median and interquartile range (IQR). We used t-tests to compare normally distributed variables and the Mann–Whitney U test to compare non-normally distributed variables (body mass index and hospital days at initial IVIG) between the groups with and without AARF.

Univariate and multivariate logistic regression analyses were performed to identify the factors associated with AARF in patients with KD. Dependent variables in the multivariable analysis were age, sex, BMI, hospital days of illness at initial IVIG, atypical KD or not, type of hospital, complex chronic conditions, transportation by ambulance, Japan Coma Scale score at admission, transportation from other hospitals, and hospital volume. Results were presented as odds ratios (ORs) and 95% confidence intervals (CIs).

We compared the proportion of coronary artery abnormalities (CAAs), IVIG resistance, medical costs, and length of stay between patients with and without AARF. CAAs were identified with a recorded diagnosis of CAAs (ICD-10 code: I254) or text data of CAAs in Japanese. IVIG resistance was defined as IVIG use at a total dose of ≥ 4.0 g/kg or a combination of any steroid, infliximab, cyclosporine, or plasma exchange that was not performed simultaneously with the initial IVIG treatment. We also performed multivariable logistic regression analyses for CAA and IVIG resistance and multiple regression analyses for the length of hospital stay and medical cost. The adjusted variables were age, sex, BMI, hospital days at initial IVIG, atypical KD, type of hospital, complex chronic conditions, transportation by ambulance, Japan Coma Scale at admission, transportation from other hospitals, and hospital volume.

For AARF patients with acute KD, we also examined whether the following treatments were performed during hospitalization: surgery (open reduction of cervical spine dislocation [Japanese surgical code: K118], dynamic stabilization of the spine [K141-3], and spinal decompression with or without fusion [K142]). Conservative treatment (cervical collar [Japanese treatment codes: J200], cervical traction [J118], halo-vest orthosis [K144], and closed reduction of the cervical spine [K117-2]). The patients' backgrounds in the surgery and conservative treatment groups were compared.

A two-sided p < 0.05 was considered significant. All statistical analyses were conducted using the Stata software (version 17.0; StataCorp LP, College Station, TX, USA).

Of the 71,913 consecutive patients with acute KD, 166 had AARF (0.23%). Forty-eight patients were not diagnosed with AARF but received conservative treatment. Demographic characteristics are shown in Table 1. Compared to those without AARF, KD patients with AARF were older, heavier, and higher in age. Furthermore, there were significant differences in the number of hospital days at initial IVIG, the proportion of atypical KD, and hospital volume.

Table 1

Descriptive characteristics of with non-AARF and AARF patients with KD
Variables	Non-AARF	AARF	p-value
	(n = 71,747)	(n = 166)
Age, mean (SD), years	2.2 (2.1)	5.0 (2.2)	< 0.001
Male (%)	41,285 (57.5%)	80 (48.2%)	0.015
Weight, mean (SD), kg	12.3 (4.9)	17.9 (5.6)	< 0.001
Height, mean (SD), cm	76.5 (34.0)	92.9 (39.0)	< 0.001
Body mass index, kg/m², median (IQR)	15.8 (14.8–17.0)	14.9 (14.0-16.1)	< 0.001
Hospital days of illness at initial IVIG, median (IQR)	2.0 (1.0–2.0)	2.0 (1.0–3.0)	< 0.001
Atypical Kawasaki Disease	3,429 (4.8%)	14 (8.4%)	0.028
Type of hospital
Academic hospital	11,500 (16.0%)	28 (16.9%)	0.96
Complex Chronic Conditions
0–1	68,911 (96.0%)	156 (94.0%)	0.17
2 ≤	2,836 (4.0%)	10 (6.0%)
Transportation by ambulance	1,760 (2.5%)	3 (1.8%)	0.59
Japan Coma Scale
Alert	71,096 (99.1%)	163 (98.2%)	0.22
Not Alert	651 (0.9%)	3 (1.8%)
Transportation from other hospital	54,486 (76.0%)	132 (79.5%)	0.29
Hospital volume
Low (1.0–29.7)	22,992 (32.0%)	31 (18.7%)	< 0.001
Middle (29.8–56.2)	24,289 (33.9%)	55 (33.1%)
Hight (≥ 56.3)	24,466 (34.1%)	80 (48.2%)
AARF, atlantoaxial rotatory fixation; IQR, interquartile range; SD, standard deviation.

In the multivariate analysis, age, BMI, and atypical KD were independently associated with AARF (Table 2). AARF was not associated with CAA or IVIG resistance. AARF was significantly associated with higher medical costs and longer lengths of hospital stay (Table 3).

Table 2

Factors associated with atlantoaxial rotatory fixation
Variables	Univariable logistic regression analyses					Multivariable logistic regression analysis
	Odds Ratio	95% confidence interval			p-value	Odds Ratio	95% confidence interval			p-value
Age, years	1.25	1.21	-	1.30	< 0.001	1.24	1.19	-	1.29	< 0.001
Male	0.69	0.51	-	0.93	0.016	0.80	0.57	-	1.11	0.182
BMI	0.89	0.82	-	0.96	0.004	0.89	0.82	-	0.96	0.004
Hospital days of illness at initial IVIG	1.02	1.00	-	1.03	0.011	1.01	0.99	-	1.04	0.251
Atypical Kawasaki Disease	1.84	1.06	-	3.18	0.03	1.95	1.12	-	3.40	0.018
Complex Chronic Conditions	1.56	0.82	-	2.96	0.175	0.92	0.41	-	2.04	0.829
Transportation by ambulance	0.73	0.23	-	2.29	0.592	0.44	0.11	-	1.82	0.258
Japan Coma Scale	2.01	0.64	-	6.31	0.232	2.56	0.79	-	8.28	0.116
Transportation from another hospital	1.23	0.84	-	1.79	0.287	1.05	0.70	-	1.57	0.807
Hospital volume
Low (1.0–29.7)
Middle (29.8–56.2)	1.68	1.08	-	2.61	0.021	1.60	1.01	-	2.55	0.047
High (≥ 56.3)	2.43	1.60	-	3.67	< 0.001	1.98	1.27	-	3.10	0.003
BMI: body mass index, IVIG: intravenous immunoglobulin

Table 3

Univariable and multivariable analyses of the association between KD with AARF and the outcomes
Variables	Univariable analyses					Multivariable analyses^*
Variables		95% confidence interval			p-value		95% confidence interval			p-value
Coronary artery abnormalities, OR^†	0.60	0.19	-	1.87	0.374	0.73	0.23	-	2.32	0.599
Intravenous immunoglobulin resistance, OR^†	1.25	0.91	-	1.72	0.166	1.05	0.74	-	1.49	0.773
Total cost, USD^‡,	3786	3080	-	4491	< 0.001	1064	346	-	1781	0.004
Length of stay, day^‡,	5.36	4.12	-	6.60	< 0.001	3.05	1.74	-	4.36	< 0.001
USD: United States Dollar
* Adjusted variables were age, sex, BMI, hospital days at initial IVIG, atypical KD, type of hospital, complex chronic conditions, transportation by ambulance, Japan Coma Scale at admission, transportation from other hospitals, and hospital volume.
† The results were analyzed using multivariable logistic regression analyses.
‡ Results were analyzed using multiple regression.

Overall, 103 (62%) patients underwent conservative treatment (Table 4). Of these, 65 (63%) underwent cervical traction. None of the patients underwent surgery. The remaining 63 patients did not receive any specific treatment for AARFs. There was no significant difference in patient background, except for sex, between the two groups with and without conservative treatment. Men were more likely to undergo conservative treatment than women.

Table 4

Patient backgrounds of KD patients with AARF divided by those with and without conservative treatment.
Variables	No treatment	Conservative treatment	p-value
	(n = 63)	(n = 103)
Age, mean (SD), years	4.7 (2.2)	5.2 (2.2)	0.17
Male (%)	38 (60.3%)	42 (40.8%)	0.014
Weight, mean (SD), kg	17.3 (4.8)	18.3 (6.1)	0.23
Height, mean (SD), cm	96.4 (31.8)	90.8 (42.8)	0.37
Body mass index, kg/m2, median (IQR)	15.1 (14.1–16.2)	14.8 (13.9–16.0)	0.38
Hospital days of illness at initial IVIG, median (IQR)	2.0 (1.0–3.0)	2.0 (1.0–3.0)	0.90
Atypical Kawasaki Disease	6 (9.5%)	8 (7.8%)	0.69
Type of hospital
Academic hospital	13 (20.6%)	15 (14.6%)	0.49
Complex Chronic Conditions
0–1	59 (93.7%)	97 (94.2%)	0.89
2 ≤	4 (6.3%)	6 (5.8%)
Transportation by ambulance	1 (1.6%)	2 (1.9%)	0.87
Japan Coma Scale
Alert	63 (100.0%)	100 (97.1%)	0.17
Not Alert	0 (0.0%)	3 (2.9%)
Transportation from other hospital	47 (74.6%)	85 (82.5%)	0.22
Hospital volume
Low (1.0–29.7)	15 (23.8%)	16 (15.5%)	0.19
Middle (29.8–56.2)	23 (36.5%)	32 (31.1%)
High (≥ 56.3)	25 (39.7%)	55 (53.4%)
AARF, atlantoaxial rotatory fixation; IQR, interquartile range; SD, standard deviation.

The present study showed the clinical features of AARF in KD patients using a national inpatient database. This study showed that older, thinner, and atypical KD patients were more likely to develop AARF.

Non-traumatic AARF can occur following cervical inflammation (such as upper respiratory tract inflammation, pharyngitis, and mumps) or otolaryngological surgery. Although the pathogenic mechanism is unclear, children generally have higher ligament laxity in the atlantoaxial joint. Inflammation of the neck may cause spasms in the cervical muscles. AARF is rare in adults and is more likely to occur in children [3, 7].

So far, only a few studies have included case reports of KD complicated by AARF [12, 17, 18]. We could report for the first time the characteristics of KD patients with AARF in a large data set. The average age of KD patients with AARF was higher than that of KD patients without AARF. Children with KD often develop cervical lymphadenitis. AARFs may be associated with cervical lymphadenitis due to KD [19].

Patients with atypical KD often develop cervical lymphadenopathy [20, 21] but do not meet the symptom criteria for KD. Patients with atypical KD may have an increased incidence of AARF due to susceptibility to delayed diagnosis of KD. Previous studies reported that AARF associated with atypical KD required prompt and appropriate diagnosis because irreversible severe deformity of the atlantoaxial joint may occur in children and require complex surgical treatment [22, 23]. In febrile patients with cervical lymphadenopathy, when the possibility of infection has been ruled out, confirming the diagnosis of KD and the early administration of IVIG may help prevent the progression of AARF.

In this study, female sex was not a risk factor in the multivariable analysis; however, there was a higher proportion of women in the univariate analysis. Although KD is considered a male-dominated disease [14, 24], previous case reports have shown that a high proportion of KD patients with AARF are women [12, 17, 18]. As women are more likely to have joint laxity than men, the proportion of AARF is thought to be higher in women [25].

This study showed that the proportion of CAA and IVIG resistance in KD with AARF was not high, but medical costs and length of stay were high and long, respectively, in KD patients with AARF. The results of this study suggest that the association between AARF and KD severity is low. A previous small study did not report any severe complications of KD [12]. KD patients with complications of AARF may have received additional orthopedic treatment, including cervical traction, resulting in longer hospital stays and higher medical costs.

To the best of our knowledge, there have been no reports of KD patients with AARFs requiring surgical intervention. However, several studies reported that pediatric patients with AARF required surgical treatments [5, 6]. Because delays in initiating treatment may result in poor prognoses, the collaboration between pediatric and orthopedic departments is necessary.

Pediatricians should be mindful of the possibility of developing AARF during the clinical course of KD. If a patient with KD develops AARF, it would be important to collaborate with orthopedic surgeons to initiate the necessary treatment promptly because AARF patients sometimes require surgical treatment. However, no AARF patients required surgical treatment in this study.

Limitations

Our study has several limitations. First, our database lacked data on the symptoms, laboratory findings, and fever duration in patients with acute KD. We adjusted the severity of KD for the state of consciousness and comorbidities on admission; however, this adjustment may have been inadequate. Second, the severity of AARF could not be adjusted for the clinical manifestations of lymphadenopathy, and the Fielding classification is unknown. Finally, patients with AARF were extracted according to the diagnosis of AARF and orthopedic treatment; however, there may have been more KD patients with complications of AARF.

Although AARF is a rare complication in patients with acute KD, it should be considered in relatively old children with atypical KD and cervical symptoms.

AARF: atlantoaxial rotatory fixation; ADL: activities of daily living; ASD: absolute standardized difference; BMI: body mass index; CAA: coronary artery abnormalities; CI: confidence interval; IQR: interquartile range; IVIG: intravenous immunoglobulin, KD: Kawasaki Disease; SD: standard deviation; USD: United States Dollar

Funding/Support: This work was supported by grants from the Ministry of Health, Labour and Welfare, Japan (21AA2007 and 20AA2005) and the Ministry of Education, Culture, Sports, Science and Technology, Japan (20H03907).

Conflict of Interest Disclosures: All authors have no potential conflicts of interest to disclose.

Availability of data and material: The datasets analyzed during the current study are not publicly available due to contracts with the hospitals providing data to the database.

Code availability: This study was analyzed using the standard packages of the Stata software version 17.0 (StataCorp LP, College Station, TX, USA).

Author contributions:

Nobuaki Michihata wrote the first draft of the manuscript. Takanori Suzuki, Akira Honda, and Itsuhiro Oka conceptualized and designed the study, drafted the initial manuscript, and reviewed and revised the manuscript. Kazuyoshi Saito, Tetsushi Yoshikawa, Hiroki Matsui, Kiyohide Fushimi, and Hideo Yasunaga conceptualized and designed the study, and coordinated and critically reviewed the manuscript for important intellectual content. All authors approved the final manuscript as submitted and agree to be accountable for all aspects of the work.

Ethics approval:

The present study was approved by the Institutional Review Board of The University of Tokyo (approval number: 3501-(3); 25 December 2017).

Consent to participate:

The requirement for informed consents was waived because of the anonymous nature of the data.

Consent for publication:

This paper isn’t included an individual’s data or image.

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Clinical features of Kawasaki disease with atlantoaxial rotatory fixation

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