Abstract
Whoo** cough is a well-known contagious disease caused by the Gram-negative bacterium Bordetella pertussis. The pathogenesis of whoo** cough is caused by various toxins including pertussis toxin. Despite widespread vaccination, the prevalence among adolescents and adults is increasing. The precise pathogenic mechanism of whoo** cough remains unknown. Cough hypersensitivity syndrome recognized as a cause of chronic cough arises from a hypersensitivity of airway sensory nerves and occurs after an inflammatory process or bacterial infection. We report a case of cough hypersensitivity syndrome following whoo** cough in a patient with genetic hypohaptoglobinemia. A 15-year-old Japanese boy was referred to our hospital with intractable coughing lasting almost 2 months. We diagnosed probable whoo** cough by a single Yamaguchi agglutinin titer of 1:1280 as well as a fourfold increase in paired blood samples. His paroxysmal cough with post-tussive emesis was consistent with the clinical feature of whoo** cough. Various treatments against known causes of chronic cough were ineffective in relieving his coughing. Therefore, we diagnosed his coughing with cough hypersensitivity syndrome following whoo** cough. Examination of his immunological status revealed genetic hypohaptoglobinemia. The cough was gradually alleviated along with restoration of the haptoglobin level, but it took almost 2 years for the coughing to be resolved completely. We monitored his hypohaptoglobinemia and cytokine profiles and found hypercytokinemia which showed autoimmune feature. Haptoglobin might have been involved in the cough hypersensitivity syndrome following whoo** cough through cytokine modification.
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Acknowledgements
We thank Professor Yoshiro Koda (Department of Forensic Medicine, Kurume University School of Medicine, Japan) for evaluating haptoglobin gene mutations, and Professor. Jan L. Ceuppens (Catholic University of Leuven, Belgium) for comments on the presented case. We thank Cathel Kerr, BSc, PhD, from Edanz Group (https://en-author-services.edanzgroup.com/) for editing a draft of this manuscript.
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KH is the corresponding author, and contributed to the collection and analysis of clinical data and writing of the manuscript, and treatment of the patient described in this report.
YK participated in discussions about this case and preparation of the manuscript.
IM contributed to the treatment decision-making for this case.
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All procedures performed in this case report were approved by the Ethics Committee of Kokura Medical Center and in accordance with the ethical standards of the institutional and/or national research committee and with the 1964 Helsinki declaration and its later amendments or comparable ethical standards.
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Hidaka, K., Kawanami, Y. & Mizuguchi, I. Cough Hypersensitivity Syndrome Following Whoo** Cough Showing Persistent Hypercytokinemia with Autoimmune Feature in Relation to Genetic Hypohaptoglobinemia: a Case Report. SN Compr. Clin. Med. 4, 159 (2022). https://doi.org/10.1007/s42399-022-01235-9
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DOI: https://doi.org/10.1007/s42399-022-01235-9