The psychological burden of an initially unexplained illness: patients with sternocostoclavicular hyperostosis before and after delayed diagnosis.

Willem A Van Der Kloot,Femke Mc Den Dulk,Neveen At Hamdy,Sadhna A Chotkan,Laurian Cs Hafkemeijer,Arnold Ap Van Emmerik,Ad A Kaptein

doi:10.1186/1477-7525-8-97

Abstract

BackgroundSternocostoclavicular hyperostosis (SCCH) is a rare, debilitating, chronic inflammatory disorder of the anterior chest wall due to a chronic sterile osteomyelitis of unknown origin. SCCH is largely underdiagnosed and often misdiagnosed. In individual cases it can remain unrecognized for years. The purpose of this study is twofold. Firstly, to evaluate the psychological condition of SCCH patients, both in the sometimes quite extended pre-diagnostic period between first manifestations and confirmed diagnosis of the disease, and in the current situation. Secondly, to investigate the relationships between the pre-diagnostic and the current psychological conditions of confirmed SCCH patients.MethodsStructured interviews were held with 52 confirmed SCCH patients. Questionnaires were included to assess posttraumatic stress symptoms, social support, aspects of pain, illness perceptions, self-reported health status, and quality of life.ResultsSCCH patients reported stronger posttraumatic stress symptoms, more unfavorable illness perceptions, lower health status, and poorer quality of life than healthy individuals and patients with other diseases or traumatic experiences. Psychological distress in the pre-diagnostic period was associated with unfavorable conditions in the current situation.ConclusionSCCH is an illness with serious psychological consequences. Psychological monitoring of patients with unexplained complaints is recommended as long as a diagnosis has not been reached.

Highlights

Sternocostoclavicular hyperostosis (SCCH) is a rare, debilitating, chronic inflammatory disorder of the anterior chest wall due to a chronic sterile osteomyelitis of unknown origin
Our first objective was to evaluate the psychological condition of SCCH patients and the extent and seriousness of their problems both in the interval between first manifestations and diagnosis of the disease, and in the current situation, after the diagnosis had been established for some time
In a similar way we found that the rating scales measuring despair, drama, concern, fright, and depression could be reduced to one factor that explained 76.4% of their variance

Summary

Introduction

Sternocostoclavicular hyperostosis (SCCH) is a rare, debilitating, chronic inflammatory disorder of the anterior chest wall due to a chronic sterile osteomyelitis of unknown origin. To evaluate the psychological condition of SCCH patients, both in the sometimes quite extended prediagnostic period between first manifestations and confirmed diagnosis of the disease, and in the current situation. Lack of awareness of the clinical manifestations of a rare disease may lead to diagnostic delay, failure of diagnosis, and misdiagnosis. These problems have been documented in a detailed report by the European Organisation of Rare Diseases, which was. The EURORDIS findings are paralleled by the results from a study of our own research group on patients with sternocostoclavicular hyperostosis (SSCH), a rare inflammatory disease of the axial skeleton [3]. In 23% the correct diagnosis was only established after the patients obtained information from sources outside their regular medical circuit

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