31P-MRS of skeletal muscle is not a sensitive diagnostic test for mitochondrial myopathy
Tina Dysgaard Jeppesen1, Bjørn Quistorff, Flemming Wibrand
1Neuromuscular Research Unit, Section 7611, National University Hospital, Rigshospitalet, Blegdamsvej 9, 2100, Copenhagen, Denmark. dysgaard@rh.dk
Abstract:
Clinical phenotypes of persons with mitochondrial DNA (mtDNA) mutations vary considerably. Therefore, diagnosing mitochondrial myopathy (MM) patients can be challenging and warrants diagnostic guidelines. (31)phosphorous magnetic resonance spectroscopy ((31)P-MRS) have been included as a minor diagnostic criterion for MM but the diagnostic strength of this test has not been compared with that of other commonly used diagnostic procedures for MM. To investigate this, we studied seven patients with single, large-scale deletions-, nine with point mutations of mtDNA and 14 healthy subjects, who were investigated for the following: 1) (31)P-MRS of lower arm and leg muscles before and after exercise, 2) resting and peak-exercise induced increases of plasma lactate, 3) muscle morphology and -mitochondrial enzyme activity, 4) maximal oxygen uptake (VO(2max)), 5) venous oxygen desaturation during handgrip exercise and 6) a neurological examination. All MM patients had clinical symptoms of MM, > 2% ragged red fibers in muscle, and impaired oxygen desaturation during handgrip. Fourteen of 16 patients had impaired VO(2max), 10/16 had elevated resting plasma lactate, and 10/11 that were investigated had impaired citrate synthase-corrected complex I activity. Resting PCr/P(i) ratio and leg P(i) recovery were lower in MM patients vs. healthy subjects. PCr and ATP production after exercise were similar in patients and healthy subjects. Although the specificity for MM of some (31)P-MRS variables was as high as 100%, the sensitivity was low (0-63%) and the diagnostic strength of (31)P-MRS was inferior to the other diagnostic tests for MM. Thus, (31)P-MRS should not be a routine test for MM, but may be an important research tool.
Insights
Diagnosing mitochondrial myopathy (MM) is challenging. While (31)phosphorous magnetic resonance spectroscopy ((31)P-MRS) shows high specificity, its low sensitivity makes it inferior to other tests for MM diagnosis.
Area of Science:
- Neurology
- Biochemistry
- Genetics
Background:
- Mitochondrial myopathy (MM) diagnosis is complex due to varied clinical presentations.
- Diagnostic guidelines are needed for MM.
- (31)phosphorous magnetic resonance spectroscopy ((31)P-MRS) is a minor diagnostic criterion for MM.
Purpose of the Study:
- To compare the diagnostic strength of (31)P-MRS with other common MM diagnostic procedures.
- To evaluate the utility of (31)P-MRS in diagnosing MM.
Main Methods:
- Studied 16 MM patients (deletions/point mutations) and 14 healthy controls.
- Utilized (31)P-MRS, plasma lactate measurements, muscle biopsy, VO(2max) testing, venous oxygen saturation, and neurological examination.
- Assessed muscle and systemic physiological responses before and after exercise.
Main Results:
- MM patients exhibited clinical symptoms, ragged red fibers, and impaired oxygen desaturation.
- Other tests revealed impaired VO(2max), elevated lactate, and reduced enzyme activity in most MM patients.
- (31)P-MRS showed high specificity but low sensitivity for MM, with impaired resting PCr/Pi ratio and leg Pi recovery.
Conclusions:
- (31)P-MRS is less effective than other methods for routine MM diagnosis.
- Despite low sensitivity, (31)P-MRS remains a valuable research tool for MM.
- MM diagnosis requires a comprehensive approach combining clinical, biochemical, and physiological assessments.
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