Literature DB >> 24470648

Periodic fever in MVK deficiency: a patient initially diagnosed with incomplete Kawasaki disease.

Valtyr S Thors1, Sebastiaan J Vastert, Nico Wulffraat, Annet van Royen, Joost Frenkel, Monique de Sain-van der Velden, Tom J de Koning.   

Abstract

Mevalonate kinase deficiency (MKD) is a rare autosomal recessive disorder causing 1 of 2 phenotypes, hyperimmunoglobulin D syndrome and mevalonic aciduria, presenting with recurrent fever episodes, often starting in infancy, and sometimes evoked by stress or vaccinations. This autoinflammatory disease is caused by mutations encoding the mevalonate kinase (MVK) gene and is classified in the group of periodic fever syndromes. There is often a considerable delay in the diagnosis among pediatric patients with recurrent episodes of fever. We present a case of an 8-week-old girl with fever of unknown origin and a marked systemic inflammatory response. After excluding infections, a tentative diagnosis of incomplete Kawasaki syndrome was made, based on the finding of dilated coronary arteries on cardiac ultrasound and fever, and she was treated accordingly. However, the episodes of fever recurred, and alternative diagnoses were considered, which eventually led to the finding of increased excretion of mevalonic acid in urine. The diagnosis of MKD was confirmed by mutation analysis of the MVK gene. This case shows that the initial presentation of MKD can be indistinguishable from incomplete Kawasaki syndrome. When fever recurs in Kawasaki syndrome, other (auto-)inflammatory diseases must be ruled out to avoid inappropriate diagnostic procedures, ineffective interventions, and treatment delay.

Entities:  

Keywords:  Kawasaki syndrome; auto inflammatory disease; fever of unknown origin; hyperimmunoglobulin D syndrome (HIDS); mevalonate kinase; mevalonic acid

Mesh:

Year:  2014        PMID: 24470648     DOI: 10.1542/peds.2012-1372

Source DB:  PubMed          Journal:  Pediatrics        ISSN: 0031-4005            Impact factor:   7.124


  6 in total

Review 1.  Treatment of hyperimmunoglobulinemia D syndrome with biologics in children: review of the literature and Finnish experience.

Authors:  Svetlana Kostjukovits; Liisa Kalliokoski; Kaisu Antila; Matti Korppi
Journal:  Eur J Pediatr       Date:  2015-02-27       Impact factor: 3.183

2.  Forme Fruste of HLH (haemophagocytic lymphohistiocytosis): diagnostic and therapeutic challenges.

Authors:  Susumu Inoue; Chetna Mangat; Yaseen Rafe'e; Mahesh Sharman
Journal:  BMJ Case Rep       Date:  2015-01-29

3.  Novel mutation identified in severe early-onset tumor necrosis factor receptor-associated periodic syndrome: a case report.

Authors:  Suhas M Radhakrishna; Amy Grimm; Lori Broderick
Journal:  BMC Pediatr       Date:  2017-04-20       Impact factor: 2.125

4.  Using the Electronic Medical Record to Correlate Kawasaki Disease Phenotypes With Clinical Outcomes.

Authors:  Preeti Jaggi; Michelle Grcic; John Kovalchin; Carolyn M Wilhelm; Cagri Yildirim-Toruner; Karen Texter
Journal:  J Pediatric Infect Dis Soc       Date:  2018-05-15       Impact factor: 3.164

5.  Appendicitis as a Leading Manifestation of Kawasaki Disease in Older Children.

Authors:  Ya-Ning Huang; Lu-Hang Liu; Jeng-Jung Chen; Yu-Lin Tai; Yih-Cherng Duh; Chien-Yu Lin
Journal:  Children (Basel)       Date:  2022-02-02

Review 6.  Real-Life Indications of Interleukin-1 Blocking Agents in Hereditary Recurrent Fevers: Data From the JIRcohort and a Literature Review.

Authors:  Caroline Vinit; Sophie Georgin-Lavialle; Aikaterini Theodoropoulou; Catherine Barbier; Alexandre Belot; Manel Mejbri; Pascal Pillet; Jana Pachlopnik; Sylvaine Poignant; Charlotte Rebelle; Andreas Woerner; Isabelle Koné-Paut; Véronique Hentgen
Journal:  Front Immunol       Date:  2021-11-11       Impact factor: 7.561

  6 in total

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