PMID- 31055642
OWN - NLM
STAT- MEDLINE
DCOM- 20200310
LR  - 20200310
IS  - 1437-160X (Electronic)
IS  - 0172-8172 (Linking)
VI  - 39
IP  - 8
DP  - 2019 Aug
TI  - Polymyositis with mitochondrial pathology or atypical form of sporadic inclusion 
      body myositis: case series and review of the literature.
PG  - 1459-1466
LID - 10.1007/s00296-019-04314-8 [doi]
AB  - Polymyositis with mitochondrial pathology (PM-Mito) is a rare form of idiopathic 
      inflammatory myopathy with no definite diagnostic criteria and similarities to 
      both PM and sporadic inclusion body myositis (s-IBM). The aim of this study is to 
      address the dilemma of whether PM-Mito is a subtype of inflammatory myopathy or 
      represents a disease falling into the spectrum of s-IBM. Herein, we report four 
      female patients diagnosed with PM-Mito, highlighting their rather atypical 
      clinical and histopathological characteristics that seem to indicate a diagnosis 
      away from s-IBM. Muscle weakness was rather proximal and symmetrical and lacked 
      the selective pattern observed in s-IBM. Patients had large-scale deletions in 
      mtDNA, reflecting the mitochondrial component in the pathology of the disease. 
      Conclusively, our study adds to the limited data in the literature on whether 
      PM-Mito is a distinct form of myositis or represents a prodromal stage of s-IBM. 
      Although the latter seems to be supported by a substantial body of evidence, 
      there are, however, important differences, such as the different patterns of 
      muscle weakness, and the good response to treatment observed in some patients. 
      Larger-scale studies are certainly needed to clarify pathogenesis and clinical 
      characteristics of PM-Mito patients, especially in therapeutic and prognostic 
      terms.
FAU - Papadimas, George K
AU  - Papadimas GK
AUID- ORCID: 0000-0003-0163-4091
AD  - 1st Department of Neurology, Medical School, Eginition Hospital, National and 
      Kapodistrian University of Athens, 74, Vas. Sophias Ave, 11528, Athens, Greece. 
      gkpapad@yahoo.gr.
FAU - Kokkinis, Charalampos
AU  - Kokkinis C
AUID- ORCID: 0000-0002-4430-4255
AD  - Department of Medical Genetics, Medical School, St. Sofia's Children's Hospital, 
      National and Kapodistrian University of Athens, Athens, Greece.
FAU - Xirou, Sophia
AU  - Xirou S
AUID- ORCID: 0000-0001-8222-091X
AD  - 1st Department of Neurology, Medical School, Eginition Hospital, National and 
      Kapodistrian University of Athens, 74, Vas. Sophias Ave, 11528, Athens, Greece.
FAU - Chrysanthou, Margarita
AU  - Chrysanthou M
AUID- ORCID: 0000-0001-5342-4585
AD  - 1st Department of Neurology, Medical School, Eginition Hospital, National and 
      Kapodistrian University of Athens, 74, Vas. Sophias Ave, 11528, Athens, Greece.
FAU - Kararizou, Evangelia
AU  - Kararizou E
AUID- ORCID: 0000-0003-3616-4552
AD  - 1st Department of Neurology, Medical School, Eginition Hospital, National and 
      Kapodistrian University of Athens, 74, Vas. Sophias Ave, 11528, Athens, Greece.
FAU - Papadopoulos, Constantinos
AU  - Papadopoulos C
AUID- ORCID: 0000-0001-7633-5032
AD  - 1st Department of Neurology, Medical School, Eginition Hospital, National and 
      Kapodistrian University of Athens, 74, Vas. Sophias Ave, 11528, Athens, Greece.
LA  - eng
PT  - Case Reports
PT  - Journal Article
PT  - Review
DEP - 20190504
PL  - Germany
TA  - Rheumatol Int
JT  - Rheumatology international
JID - 8206885
RN  - 0 (DNA, Mitochondrial)
RN  - 0 (Glucocorticoids)
RN  - 0 (Immunosuppressive Agents)
SB  - IM
MH  - Adult
MH  - Aged
MH  - Biopsy
MH  - DNA, Mitochondrial/genetics
MH  - Diagnosis, Differential
MH  - Female
MH  - Gene Deletion
MH  - Glucocorticoids/therapeutic use
MH  - Humans
MH  - Immunosuppressive Agents/therapeutic use
MH  - Middle Aged
MH  - Mitochondria, Muscle/drug effects/genetics/*pathology
MH  - Muscle Weakness
MH  - Muscle, Skeletal/drug effects/*pathology/physiopathology
MH  - Myositis, Inclusion Body/genetics/*pathology/physiopathology
MH  - Polymyositis/drug therapy/genetics/*pathology/physiopathology
MH  - Predictive Value of Tests
MH  - Treatment Outcome
OTO - NOTNLM
OT  - Inclusion body myositis
OT  - Mitochondria
OT  - Polymyositis
OT  - mtDNA
EDAT- 2019/05/06 06:00
MHDA- 2020/03/11 06:00
CRDT- 2019/05/06 06:00
PHST- 2018/12/11 00:00 [received]
PHST- 2019/04/26 00:00 [accepted]
PHST- 2019/05/06 06:00 [pubmed]
PHST- 2020/03/11 06:00 [medline]
PHST- 2019/05/06 06:00 [entrez]
AID - 10.1007/s00296-019-04314-8 [pii]
AID - 10.1007/s00296-019-04314-8 [doi]
PST - ppublish
SO  - Rheumatol Int. 2019 Aug;39(8):1459-1466. doi: 10.1007/s00296-019-04314-8. Epub 
      2019 May 4.