Vol.:(0123456789) 1 3
Rheumatology International
https://doi.org/10.1007/s00296-019-04478-3
COHORT STUDIES
Pattern and diagnostic evaluation of systemic autoinfammatory
diseases other than familial Mediterranean fever among Arab
children: a multicenter study from the Pediatric Rheumatology Arab
Group (PRAG)
Sulaiman M. Al‑Mayouf
1
· Abdulaziz Almutairi
1
· Safya Albrawi
2
· Basil M. Fathalla
3
· Raed Alzyoud
5
·
Abdullatif AlEnazi
4
· Mohammed Abu‑shukair
5
· Adel Alwahadneh
5
· Abdullah Alsonbul
1
·
Mabruka Zlenti
6
· Ebtisam Khawaja
6
· Awatif Abushhaiwia
6
· Khulood Khawaja
7
· Zakiya AlMosawi
8
·
Wafa Madan
8
· Muna Almuatiri
9
· Nora Almuatiri
10
· for Pediatric Arab Rheumatology Group (PRAG)
Received: 8 August 2019 / Accepted: 8 November 2019
© Springer-Verlag GmbH Germany, part of Springer Nature 2019
Abstract
To defne the spectrum and phenotypic characteristics of systemic autoinfammatory diseases (SAIDs) other than familial
Mediterranean fever (FMF) in Arab children and to delineate diagnostic evaluation. Data retrospectively collected on patients
with clinical and/or genetically proven SAIDs other than FMF at 10 tertiary Arab pediatric rheumatology clinics from 1990
to 2018. The collected data comprised the clinical fndings and diagnostic evaluation including genetic testing, the provided
treatment and the accrual damage related to SAIDs. A total of 144 patients (93 female) with a median age at onset of 2.5
(range 0.1–12) years were enrolled. The initial diagnosis was inaccurate in 49.3%. Consanguinity rate among parents was
74.6%. The median time-to-diagnosis for all SAIDs was 2.5 (range 0.1–10) years. There were 104 patients (72.2%) with a
confrmed diagnosis and 40 patients with suspected SAIDs. Seventy-two had monogenic and 66 patients with multifactorial
SAIDs while six patients had undiferentiated SAIDs. The most frequent monogenic SAIDs were LACC1 mediated mono-
genic disorders (n = 23) followed by CAPS (12), TRAPS (12), HIDS (12), and Majeed’s syndrome (6). The most frequent
multifactorial SAIDs was CRMO (34), followed by PFAPA (18), and early onset sarcoidosis (EOS) (14). Genetic analysis was
performed in 69 patients; 50 patients had genetically confrmed disease. Corticosteroid used for 93 patients while biologic
agents for 96 patients. Overall, growth failure was the most frequent accrual damage (36%), followed by cognitive impair-
ment (13%). There were three deaths because of infection. This study shows a heterogenous spectrum of SAIDs with a high
number of genetically confrmed monogenic diseases; notably, LACC1 associated diseases. Hopefully, this work will be the
frst step for a prospective registry for SAIDs in Arab countries.
Keywords Autoinfammatory diseases · Familial mediterranean fever · CAPS · TRAPS · LACC1 · Arabs
Rheumatology
INTERNATIONAL
* Sulaiman M. Al-Mayouf
mayouf@kfshrc.edu.sa
1
Department of Pediatrics, King Faisal Specialist Hospital
and Research Center, Alfaisal University, Po Box 3354,
Riyadh 11211, Saudi Arabia
2
Royal Hospital, Muscat, Oman
3
Al Jalila Children’s Specialty Hospital, Dubai,
United Arab Emirates
4
King Fahad Medical City, Riyadh, Saudi Arabia
5
Queen Rania Children Hospital, Amman, Jordan
6
Tripoli Children Hospital, Tripoli, Libya
7
Al-Mafraq Hospital, Abu Dhabi, United Arab Emirates
8
Salmaniya Medical Complex, Manama, Bahrain
9
Adan Hospital, Hadiya, Kuwait
10
AlSabah Hospital, Al Asimah, Kuwait