Background: Transient primary bone marrow edema syndrome (TPBMES), a set of conditions affecting weight-bearing joints, mostly of the lower limbs, presents acutely with severe local pain, disproportionate to the physical findings. This disease is designated by an array of terms, not all of which are in the consensus. But they seem to refer to a single disease entity, whether the hips, the knees, or the foot and ankle are affected, singly, bilaterally, or in a migratory pattern. The diagnosis of bone marrow edema of a temporary nature is based on the MRI. Moreover, systemic osteoporosis has been detected in association with some cases of TPBMES, but the significance of this combined occurrence is not clear. Moreover, whether "migratory" TPBMES represents a more aggressive expression of the disease is not yet settled.
Objectives: This study was initiated to resolve the numerous
ambiguities concerning this disease entity.
Data Sources: A search in PubMed looking for TPBMES or
equivalent terms.
Study Eligibility Criteria: Single or isolated case reports
were excluded. Two reviews were excluded as they did not agree with the basic
classification of the entity. Inclusion criteria of patients: a history of
sudden onset of pain in the affected joint, in the absence of more than minimal
trauma, and confirmation of bone marrow edema by MRI and follow-up evidence of
the resolution, as shown by MRI. Exclusion criteria of patients: insidious
onset of pain and evidence of avascular necrosis, infection, significant trauma
of bone, stress fractures, or inflammatory arthropathy, based on the MRI and
the laboratory. Remote studies were not based on the MRI.
Study Appraisal and Synthesis Methods: Based on a review
featuring 141 patients, several clinical, radiological and pathological data
were extracted into an all-inclusive table (not shown). Four tables were built
thereof. Tables 2-4 were conceived to summarize independently all the variables
in every recognized form of TPBMES (see Table 1). The association between
TPBMES and systemic osteoporosis is shown in an additional table.
Results: Table 2 features the demographic and clinical
studies, as distributed among the different TPBMES subtypes. A total of 546
patients were assigned mainly to 342 TOH patients, most of them males. Next,
were 105 patients with TOH of pregnancy. A lesser subset was composed of 49
F&A patients, mainly women. Table 3 shows the allocation of clinical
features and risk factors to TPBMES subsets. Pain was by far the most frequent
symptom. Subchondral fractures were detected in TOH and TOH of pregnancy.
Overweight and vitamin D deficiency have been suggested as additional risk
factors. Table 4 depicts the treatment and follow-up of TPBMES patients.
Bisphosphonates have been recommended for TOH and "migratory"
osteoporosis patients. Tables 5 and 6 demonstrate the interaction between
TPBMES and systemic osteoporosis and with migratory osteoporosis, highlighting
the common self-limited nature of TPBMES, as well as the frequently extended
course of this combined occurrence.
Limitations: See Fig. 1.
Conclusions: The different subsets in the TPBMES
classification have several features in common, notably an MRI-based diagnosis
and a self-limited outcome, even in the absence of modern therapy. Thus, they
should be considered as belonging to a single disease entity, with some
variable traits. Of note, a tendency for TPBMES to be associated with systemic
osteoporosis, sometimes in addition to a "migratory" pattern, has
been reported in conjunction with an abnormally extended course of the disease.
These are the patients who raised our concern and who evoked the necessity for
a therapeutic intervention to limit their suffering (see suggested management
protocol, Fig. 2).
Author(s) Details:
Nissim Ohana,
Department of Architecture, Jamia Millia Islamia, New Delhi, India.
Dimitri Sheinis
Department of Orthopedic Surgery, Soroka University Medical Center, and
Faculty of Health Sciences, Ben Gurion University of the Negev, Beer-Sheva,
Israel.
Daniel Benharroch
Department of Pathology, Soroka University Medical Center and Faculty of
Health Sciences, Ben Gurion University of the Negev, Beer-Sheva, Israel.
Please see the link here: https://doi.org/10.9734/bpi/nvmms/v8/115
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