Diffuse idiopathic skeletal hyperostosis in a 33-year-old woman with PCOS and metabolic syndrome: a rare scenario

Abstract

Diffuse idiopathic skeletal hyperostosis (DISH) is a common degenerative enthesopathy seen in the elderly with male preponderance. It is uncommon in patients before 50 years of age and is extremely rare in patients younger than 40 years. We report a case of 33-year-old unmarried woman who presented with inflammatory spinal pain and stiffness, limited chest expansion, decreased range of spinal motion and postural abnormalities, all of which suggested the diagnosis of ankylosing spondylitis, considering the patient’s age. But, further evaluation led us to the final diagnosis of DISH with associated metabolic syndrome and polycystic ovarian syndrome (PCOS). To the best of our knowledge, our patient is the first reported case of DISH in a woman less than 40 years of age, and also the first case of DISH associated with PCOS and metabolic syndrome.

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“…Moreover, we only found one case of DISH in a patient belonging to the 20-29-year age group [7] and the other the 30-39year age group [3]. Our case features an extremely rare case of early-stage thoracic DISH in a 24-year-old male, an age that is far below the expected age of onset for DISH symptoms.…”

“…Chitten and James (2019) [3] also presented a unique study of DISH, which involved a 33-year-old woman with polycystic ovarian syndrome (PCOS). Both reports merely used plain radiography and/or CT scan and MRI to confirm DISH, while the use of SPECT/CT was not mentioned.…”

“…It often remains undetected as patients may not experience any symptoms in the early stages until it progresses to later stages, most commonly causing stiffness, pain, and restricted range of motion in the neck or back [2]. The prevalence of DISH was reported to be higher in male populations and in patients over 50 years old, while it is extremely rare for individuals under the age of 40 to have the said condition [3]. Until the present, consensus in the medical field regarding the diagnostic criteria of the disease has not yet been achieved.…”

DISHphagia – A Riddle Unwrapped a Clinical Case with Literature Review

“…Diff use idiopathic skeletal hyperostosis (DISH) is a common but underdiagnosed systemic musculo-skeletal disease, which is characterized by calcifi cation and ossifi cation of the spinal ligaments and aff ecting generally the peripheral skeleton [8,12,13,19]. The most aff ected part of the spine is the thoracic region, followed by the lumbar and cervical ones [20].…”

“…This disorder was fi rst described in 1950 by Forestier and his student Roté s-Querol and was renamed as DISH by Resnick in 1975 [10,11]. This condition is a systemic non-infl ammatory disease, characterized by calcifi cation and ossifi cation of the spinal ligaments and it may aff ect all extraspinal locations, in which tendons and ligaments are attached to the bone [12,13].…”