Acute odontogenic infection combined with crowned dens syndrome: a case report

Abstract

Background Calcium pyrophosphate dihydrate crystal deposition disease is a condition in which calcium pyrophosphate dihydrate crystal is deposited in joint cartilage and ligaments. Calcium pyrophosphate dihydrate crystal deposition disease that involves calcification around the odontoid process of the second cervical vertebra is called crowned dens syndrome. Crowned dens syndrome is accompanied by fever in addition to acute and intense neck, posterior head, and temporal pain; thus, distinguishing crowned dens syndrome may be difficult in the presence of odontogenic infection. To the best of our knowledge, this is the first report describing a patient with crowned dens syndrome with coexisting odontogenic infection. Case presentation A 75-year-old Japanese woman was examined in the Emergency Department of this hospital due to a chief complaint of worsened buccal swelling on the left side. An odontogenic infection was considered, and she underwent her first examination. She presented with a body temperature of 37.4 °C, marked swelling and tenderness of her left lower eyelid through to her left cheek, and pain on the left temporal area. Blood tests revealed a leukocyte count of 6700/μL and a C-reactive protein level of 7.15 mg/dL. There was swelling and pain around the gingiva and acute purulent apical periodontitis of left maxillary second premolar. Cellulitis of the left cheek was diagnosed. After performing drainage of the pus, antibiotic treatment was initiated. Although her clinical symptoms improved, blood tests on day 9 of hospitalization revealed a leukocyte count of 6500/μL and a C-reactive protein level of 25.62 mg/dL, which were indicative of worsening symptoms. Computed tomography was performed to evaluate remote infection and images revealed a calcification around the odontoid process of her second cervical vertebra. When she was referred to the Orthopedic Surgery Department, pseudogout of the cervical spine was diagnosed. Subsequently, oral acetaminophen was initiated, and both her leukocyte count and C-reactive protein improved markedly. Conclusions In the presence of persistent fever and abnormally high leukocyte and C-reactive protein indicative of an inflammatory reaction, coexistence of pseudogout should be considered. In particular, when symptoms of temporal pain are present, the possibility of pseudogout of the cervical spine must be considered in the differential diagnosis.

Crowned dens syndrome: A case series of 72 patients at eight teaching hospitals in Japan

“…In our study, 10 cases of CDS occurred during hospitalization, two of which took more than 2 days to be diagnosed. Previous studies have also reported multiple cases of CDS during hospitalization for dental infection, 9 post brain surgery, 16 post endoscopic retrograde cholangiopancreatography, 22 pneumonia, 23 and after acute cerebral infarction 24 . New symptoms such as fever during hospitalization may be due to flare‐ups of the primary disease, side effects of medication, or complications of invasive procedures such as catheterization, making CDS diagnosis even more difficult than in outpatients.…”

“…It is of interest that, although the majority of CDS cases were treated with NSAIDs, acetaminophen was prescribed in as many as 1/4 of cases. Two previous CDS reports from Japan also used acetaminophen for treatment 9,20 . Acetaminophen has become the first‐line drug of choice in the management of mild chronic pain in the elderly due to its safety profile compared to NSAIDs 29 .…”

“…A case report by Oka et al summarized 69 cases from previous case reports and reported that in 50% of cases, the initial diagnosis was wrong. 5 Meanwhile, more than 30 case reports and clinical images have been published since 2010, [6][7][8][9][10][11][12][13][14] and CDS is frequently mentioned in Japanese medical books. We have the impression that we regularly diagnose and manage CDS, suggesting a perception gap between these previous studies and our clinical experience.…”

“…Meanwhile, more than 30 case reports and clinical images have been published since 2010, 6 , 7 , 8 , 9 , 10 , 11 , 12 , 13 , 14 and CDS is frequently mentioned in Japanese medical books. We have the impression that we regularly diagnose and manage CDS, suggesting a perception gap between these previous studies and our clinical experience.…”

Report of four cases of crowned dens syndrome: Clinical presentation, CT findings and treatment

“…In contrast to the gout frequently occurring in older adult males, pseudogout is more common in older adult females. Pseudogout frequently affects the knees, hands, shoulder joints, elbow joints and feet, and it occasionally occurs in the cervical, thoracic and lumbar spine (55). Pseudogout occurring around the cervical odontoid process may cause neck pain, which usually manifests as acute or subacute moderate to severe neck pain, even restricted cervical range of motion and occipital pain (25).…”