Implication of bisphosphonate use in the treatment of SAPHO syndrome: Case report and discussion of current literature

Abstract

Even though increasing knowledge is emerging about synovitis, acne, pustulosis, hyperostosis and osteitis (SAPHO) syndrome its pathogenesis remains enigmatic. Women are preferentially affected by SAPHO syndrome. Here we present the case of a 39-year-old woman suffering from this syndrome whose bone involvement was first interpreted as diffuse sclerosing osteomyelitis of the mandible. As treatment with clindamycin did not improve the symptoms, the decision was made to administer bisphosphonates intravenously. This treatment led to a rapid improvement in symptoms, which could be explained by the apparent tendency of bisphosphonates to exert a positive effect on the jaw. With this case report we attempt to offer an explanation for the influence of this group of medications on patients suffering from SAPHO syndrome with mandibular involvement.

Effect of Ibandronate Therapy on Serum Chemerin, Vaspin, Omentin-1 and Osteoprotegerin (OPG) in Postmenopausal Osteoporotic Females

“…However, there was no significant correlation between serum OPG and chemerin, vaspin, or omentin-1. It has been reported that RANKL causes activation of osteoclast, and OPG blocks this activation and suppresses the action of osteoclast, which are the main cells responsible for bone resorption ( Gorecki et al, 2015 ). Based on our findings, we propose a possible mechanism through which ibandronate affects these adipokines and prevents osteoporosis ( Figure 4 ).…”

The treatment of diffuse sclerosing osteomyelitis with oral bisphosphonates

“…In all the cases reported, bisphosphonate treatment had a beneficial effect on the patients' pain, often within a few days of commencing treatment. One case reported fever lasting 2 days related to IV zoledronate, which is a recognised side effect of the drug. The follow‐up periods ranged from 1 month to 10 years.…”

SAPHO Syndrome in an Adult Male with Ulcerative Colitis

“…5,6 Awareness of this syndrome is necessary to differentiate it from other similar conditions to prevent or avoid its misdiagnosis. 1,7 We present a case of a young male from North Pakistan, a diagnosed case of ulcerative colitis, who developed skin, osseous and articular features of SAPHO syndrome gradually with the involvement of anterior chest wall. We intend to highlight the diagnostic challenges faced in such cases.…”

“…5 At present, there is no specific treatment for SAPHO syndrome; however, symptomatic relief with first-line agents including non-steroidal anti-inflammatory drugs and corticosteroids is the main objective. 3,[5][6][7] Antibiotics have also been tried considering the detection of propionibacterium acnes in bone biopsies of few patients without any definite promising results. 5,7 Recently, inclusion of drugs like methotrexate, intra-venous pamidronate, calcitonin, bisphosphonates and antitumor necrosis factor agents in the management of this syndrome has shown reassuring results.…”

“…3,[5][6][7] Antibiotics have also been tried considering the detection of propionibacterium acnes in bone biopsies of few patients without any definite promising results. 5,7 Recently, inclusion of drugs like methotrexate, intra-venous pamidronate, calcitonin, bisphosphonates and antitumor necrosis factor agents in the management of this syndrome has shown reassuring results. 1,7 To conclude, different modalities of radiology along with clinical manifestations play a vital role in the challenging diagnosis of this syndrome affecting skin, bones and joints having overlapping features with multiple other conditions.…”