Letter to Editor - A Clinical ‘PIC- Perspectives, issues & Concerns” on Idiopathic Transient Osteoporosis during Pregnancy with alarm bells on its Idiopathic nature - Response

Dear Editor, At the outset, I would like to congratulate the authors of the article published in your journal in the current issue entitled- Idiopathic Transient Osteoporosis during Pregnancy – Report of a Clinical Case JOCR November – December 2019 Volume 9 Issue 6 Page 54-57. However, I regret to mention that the workup mentioned in this article needs to be improved, before one can come to definitive diagnosis of Idiopathic Transient Osteoporosis during Pregnancy. I would like to highlight various perspectives, issues & concerns, the knowledge of which are must for the readers of this journal pertaining to this disorder. It is pertinent to mention no doubt the increasing utilization of magnetic resonance imaging (MRI) has allowed physicians to investigate edematous changes in bone marrow, a clinically important entity which was previously undetected on conventional radiographs. The first use of the term “bone marrow edema” was by Wilson et al in 1988, and the term “bone marrow edema syndrome” was described in 1993 after the investigation of histological specimens [1]. Later on, importantly Hayes et al. advocated that the term “transient bone marrow edema syndrome” should be used for patients in whom the bone marrow edema pattern was not accompanied by radiographic evidence of osteopenia [2]. And thus separating the two entities i.e. “the transient bone marrow edema syndrome” and “Transient Osteoporosis” Occurrence of hip pain during pregnancy is quite common, although transient osteoporosis as a condition causing this symptom is uncommonly seen. Clinicians should also be aware of intra-articular disorders such as loose bodies, and labral tears; peri-articular pathology such as tendinitis and bursitis; or extra-articular conditions such as referred pain from the lumbar spine, the sacroiliac joint, and or from nerve entrapment syndromes. So, a detailed history and physical examination is helpful to narrow the differential diagnoses, which, in turn, dictate the modal

AB1028 A SINGLE-CENTER EXPERIENCE WITH TRANSIENT OSTEOPOROSIS – PATIENT CHARACTERISTIC AND APPROACH TO THERAPY.

Background:Transient osteoporosis (TOP) or transient bone marrow edema syndrome is an enigmatic condition of unknown etiology first described in pregnant women. Though usually self limited, TOP causes pain and debilitation and predisposes the patient to avascular necrosis or fractures. The course can be protracted and patient may suffer relapses. Diagnostic method of choice is magnetic resonance imaging (MRI). Based on small case series and expert opinion, several therapeutic approaches have been proposed to hasten the recovery, including bisphosphonates, calcitonin, teriparatide. However, the literature is scarce and additional experience is needed to promote the understanding of this condition.Objectives:To present our experience with TOP, including patient’s characteristic, approach to diagnosis, prognosis and therapyMethods:It is retrospective, single center study, conducted in Rambam healthcare campus, Haifa, Israel. All the medical files of patients referred to Rheumatology unit between years 2010-2020 were screened for diagnosis of TOP. Search words included: “osteoporosis”, “bone marrow edema”, “transient osteoporosis”. The files were reviewed for patient’s characteristics, modality of diagnosis, duration until full recovery, treatments and relapses.Results:Eight patients with at least one episode of TOP were identified using the search words. Six patients (75%) were female. Three female patients developed TOP during or shortly after pregnancy. Two patients – one male and one non pregnant female suffered from TOP after bariatric surgery. One pregnant woman had a strong family history of TOP. The most frequent involved site in order of frequency were: hip (4/8), ankle (3/8) and knee (2/8). Six patients presented with more than one simultaneous site of TOP (hips, knees and ankles). Blood count, liver and Kidney function tests, markers of bone resorption, rheumatoid factor, Anti cyclic citrullinated peptide, Antinuclear antibodies were negative in all of the patients. C-reactive protein was elevated in 4/8 patients, Erythrocyte sedimentation rate was elevated in 2/8 patients. All patients had vitamin D deficiency. The diagnosis was confirmed by MRI. All the patients were treated with vitamin D and intra-venous Pamidronate, one patient with addition of calcitonin and one patient with addition of intra venous Iloprost. Time to recovery ranged from 1.2 to 6 months. The time to recovery was the same in pregnancy related TOP. Recovery was confirmed with follow-up MRI in all the patients. Relapses occurred in 4/8 patients and only one them had pregnancy related TOP. All the patients were treated by multidisciplinary team, including orthopedic surgeon, physiotherapist and psychologist when needed.Conclusion:Our experience with TOP was enriched in patients presenting with more than one site of disease probably representing referral bias. Pregnancy related TOP was associated with lower risk of relapse. In terms of time to recovery there was no trend between pregnancy related and non related TOP or one site versus several sited TOP. None of the patients developed fracture, advocating in favor of adding bisphosphonates to therapy. Multidisciplinary approach is an essential part of TOP treatment strategy.Disclosure of Interests:Sami Giryes: None declared, Katya Dolnikov: None declared, Alexandra Balbir-Gurman Consultant of: Novartis, Daniela Militianu: None declared, Natalia Puchkov: None declared, Yolanda Braun-Moscovici: None declared

An Unusual Cause of Ankle Pain in a Pediatric Patient – Isolated Transient Bone Marrow Edema of the Talus - Case Report and Review of Literature

Transient osteoporosis of the talus is an uncommon condition of unknown etiology that is characterized clinically by ankle pain without a history of trauma. We present a case of isolated transient bone marrow edema of the talus in a pediatric patient which is the youngest described in the literature.

Recurrent Migratory Transient Bone Marrow Edema of the Knees Associated with Low Vitamin D and Systemic Low Bone Mineral Density: A Case Report and Literature Review

Transient bone marrow edema (TBME) is a self-limiting disease characterized by joint pain with localized bone marrow edema by MRI and has been reported in many case series and case reports. It is well known that joints of the lower extremity including hips, knees, ankles, and feet are the classical sites for TBME. Many theories have been proposed for the pathogenesis of TBME. Systemic osteopenia and vitamin D deficiency is one of the theories that have been suggested in the last few years. In this case report, we present a middle-aged male patient, who presented with 4 attacks of TBME in both knees between September 2016 and August 2017. The patient was found to have persistently low vitamin D and osteopenic T score in DXA scan of the lumbar spine and hips. Patients of TBME usually present with joint pain that is provoked by weight-bearing physical activity. The aim of this case report is to raise the awareness that TBME can be the initial presentation of systemic loss of bone mineral density.