Abstract


             
                
                   Background


                Dermatomyositis (DM) is a rare syndrome that belongs to the group of idiopathic inflammatory myopathies. The association between DM and malignancy is well recognized, and the severity of DM symptoms has been linked to the progression of metastatic disease.


             
                
                   Case presentation


                We report the case of a 42-year-old man that was diagnosed with dermatomyositis (DM) and rectal cancer. Proctectomy was performed, and DM symptoms were resolved postoperatively. One year and 9\ua0months after the surgery, liver metastasis occurred accompanied by the exacerbation of DM symptom. Partial resection of the liver was performed, and postoperative course was uneventful. DM symptoms improved postoperatively, and no evidence of cancer recurrence or DM symptoms was observed 2\ua0years after the second surgery. To date, few reports have described recurring cases of DM accompanied by colorectal cancer in detail. We reviewed four similar cases that were reported poor prognoses with treatment resistance. However, our case report demonstrates good long-term results with resection of metastatic lesion.


             
                
                   Conclusions


                It is important to check the exacerbation of DM symptoms, as this symptom sometimes preceded cancer relapse during the follow-up of our patient with DM and colorectal cancer

Atsushi Kagimoto

Kazuaki Miyamoto

Kazuhiro Toyota

Koichi Oishi

Kosuke Ono

Manabu Shimomura

Masahiro Ikeda

Masayuki Shishida

Naofumi Tsukiyama

Satoshi Shibata

Seiji Sadamoto

Tadateru Takahashi

Ono, Kosuke

Shimomura, Manabu

Toyota, Kazuhiro

Kagimoto, Atsushi

Tsukiyama, Naofumi

Shishida, Masayuki

Oishi, Koichi

Miyamoto, Kazuaki

Shibata, Satoshi

Ikeda, Masahiro

Sadamoto, Seiji

Takahashi, Tadateru

Springer OAI

Successful resection of liver metastasis detected by exacerbation of skin symptom in a patient with dermatomyositis accompanied by rectal cancer: a case report and literature review

Springer - Publisher Connector

CASE REPORT Open Access
Successful resection of liver metastasis
detected by exacerbation of skin symptom
in a patient with dermatomyositis
accompanied by rectal cancer: a case
report and literature review
Kosuke Ono1, Manabu Shimomura1*, Kazuhiro Toyota1, Atsushi Kagimoto1, Naofumi Tsukiyama1,
Masayuki Shishida1, Koichi Oishi1, Kazuaki Miyamoto1, Satoshi Shibata1, Masahiro Ikeda1, Seiji Sadamoto1
and Tadateru Takahashi1,2
Abstract
Background: Dermatomyositis (DM) is a rare syndrome that belongs to the group of idiopathic inflammatory
myopathies. The association between DM and malignancy is well recognized, and the severity of DM symptoms
has been linked to the progression of metastatic disease.
Case presentation: We report the case of a 42-year-old man that was diagnosed with dermatomyositis (DM) and
rectal cancer. Proctectomy was performed, and DM symptoms were resolved postoperatively. One year and 9 months
after the surgery, liver metastasis occurred accompanied by the exacerbation of DM symptom. Partial resection of the
liver was performed, and postoperative course was uneventful. DM symptoms improved postoperatively, and no
evidence of cancer recurrence or DM symptoms was observed 2 years after the second surgery. To date, few reports
have described recurring cases of DM accompanied by colorectal cancer in detail. We reviewed four similar cases that
were reported poor prognoses with treatment resistance. However, our case report demonstrates good long-term
results with resection of metastatic lesion.
Conclusions: It is important to check the exacerbation of DM symptoms, as this symptom sometimes preceded cancer
relapse during the follow-up of our patient with DM and colorectal cancer.
Keywords: Dermatomyositis, Colorectal cancer, Recurrence, Liver metastasis
Background
Dermatomyositis (DM) is a rare syndrome that belongs to
the group of idiopathic inflammatory myopathies. It is
characterized by heliotrope rashes, Gottron’s papules, and
proximal muscle weakness [1, 2]. The association between
DM and malignancy has been well recognized for many
years [3]. In particular, DM has been linked to tumors in
the ovaries, lungs, pancreas, and stomach; as well as non-
Hodgkin’s lymphoma, and sometimes, colorectal cancer
[4]. Malignancies are usually diagnosed within a year from
the onset of myopathy, and the extent of DM symptoms
has been associated with the progression of metastatic
diseases [5]. Recent reports have demonstrated the rela-
tionship between the exacerbation of DM symptoms and
cancer recurrence [6].
We report a case of a patient who presented with DM
and colorectal cancer, and underwent curative resection
for liver metastasis that was detected by the exacerbation
of DM symptom. We also reviewed similar reported
cases and analyzed the clinicopathological features of
* Correspondence: manabus761215@gmail.com
1Department of Surgery, National Hospital Organization Higashihiroshima
Medical Center, 513, Jike, Saijyo-cho, Higashihiroshima, Hiroshima 739-0041,
Japan
Full list of author information is available at the end of the article
© The Author(s). 2017 Open Access This article is distributed under the terms of the Creative Commons Attribution 4.0
International License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, and
reproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link to
the Creative Commons license, and indicate if changes were made.
Ono et al. Surgical Case Reports  (2017) 3:3 
DOI 10.1186/s40792-016-0281-z
patients with DM accompanied by recurrent colorectal
cancer.
Case presentation
A 42-year-old man presented with a skin rash on his
face and finger (Fig. 1). His past history included an ap-
pendectomy at 15 years old. There was no family history.
The patient was diagnosed with DM based on the pres-
ence of a heliotrope rash, Gottron’s papules, dysphagia,
proximal muscle weakness, and raised muscle-associated
enzyme levels; Jo-1 antibody was negative. He under-
went medical examinations to evaluate the existence of
accompanying malignant tumors. Colonoscopy revealed
type 2 tumors in the rectum (Fig. 2a); the patient was
diagnosed with adenocarcinoma following a biopsy.
Computed tomography (CT) scan showed no distant
metastases and some swollen lymph nodes. We per-
formed anterior resection of the rectum and regional
lymphadenectomy. The pathological diagnosis was mod-
erately differentiated adenocarcinoma, T3N1M0, and
stage III according to the 7th edition of the International
Union Against Cancer TNM classification (Fig. 2b). DM
symptoms improved after surgery. The patient received
adjuvant chemotherapy of capecitabine for 6 months,
and blood examinations were performed every 3 months
during follow-up at the outpatient clinic. No evidence of
the recurrence in the tumor marker, CT scan, and colon-
oscopy were observed 1 year postoperatively. During this
period, he did not receive immunosuppressants such as
predonisolone.
One year and 9 months after surgery, he was referred
to the hospital due to exacerbated skin symptoms,
including heliotrope rash and Gottron’s papules. The
laboratory examination revealed elevated levels of creat-
ine kinase (671 U/l), aspartate transaminase (122 IU/l),
lactate dehydrogenase (525 IU/l), and carcinoembryonic
antigens (6.5 ng/ml). Based on these findings, we
suspected rectal cancer recurrence, and the CT scan
revealed a 30-mm tumor in the segment 7 of the liver
(Fig. 2c). There were no other distant metastases in the
gastrointestinal endoscopy and colonoscopy. The patient
was diagnosed with recurring rectal cancer, and he
underwent partial resection of the liver at segment 7.
Metastatic adenocarcinoma was confirmed in the histo-
pathological examination (Fig. 2d); postoperative course
was uneventful.
The patient’s skin symptoms improved postoperatively.
Laboratory values including CPK, aldolase, and CEA also
decreased. Capecitabine was administrated as an adju-
vant chemotherapy for 6 months. No evidence of cancer
recurrence and DM symptoms were observed 2 years
after the hepatectomy.
Discussion
The association between DM and malignant tumors was
independently described nearly 100 years ago by two
investigators, Kankeleit and Stertz [7]. The incidence of
cancer in adult patients with DM is approximately 13–
42% [8]. Although its underlying pathogenesis remains
unknown, it has been considered as one of the probable
reasons why paraneoplastic condition producing bio-
active mediators causes immune reactions in the muscle
fibers and skin [5, 9]. Patients with DM and malignant
disease have reportedly a worse prognosis than patients
with idiopathic DM; surgical resection was not possible
in many of the patients with cancer because of the
advanced stage of their disease [10]. Difficulties in the
management of cancer chemotherapy and medication
for DM symptoms have also been reported due to severe
dysphagia in patients with unresectable metastases [6].
Successful treatment of these malignancies can lead to
resolution of paraneoplastic symptoms. Several reports
have shown that tumor resection can lead to the reso-
lution of paraneoplastic DM symptoms [11]. In our case,
the extent of DM symptoms was associated with the
treatment course of rectal cancer, suggesting that we
Fig. 1 a Facial erythma and heliotrope rash. b Gottron’s papules overlying knuckles
Ono et al. Surgical Case Reports  (2017) 3:3 Page 2 of 4
were dealing with a case of paraneoplastic DM caused
by rectal cancer.
It has been reported that symptoms also worsen when
the malignancy recurred after resection of the primary
disease in patients with paraneoplastic DM. However, to
date, few reports have described details of recurring
disease in the patients with DM accompanied by colo-
rectal cancer. We have therefore investigated recurrent
cases of DM accompanied by colorectal cancer to clarify
the clinical features of these cases.
A literature search was conducted in April 2016 using
the PubMed and Ichushi database to obtain English and
Japanese literature describing recurring cases of patients
with colorectal cancer and DM; four reports were found
[6, 12–14].The keywords used in the search were “derm-
atomyositis” and “colorectal cancer.” The details of clini-
copathological features of these cases, including the
present case, are demonstrated in Table 1. There were
two men and three women with a mean age of 54 years.
Three patients had colon cancer and two had rectal can-
cer. Two cases had synchronous liver metastases, and
hepatic resections were performed after the resection of
the primary tumors. Recurrence sites were in the liver in
three cases, lung in two cases, and local recurrence in
one case (including overlapped case). Four reported
cases presented moderately differentiated adenocarcin-
oma; however, the pathological differences between
general colorectal cancers were not determined. The
molecular findings for such cases were not described,
although one case presented BRAF V600E mutation
(case 3). Further investigation is needed to clarify these
findings. DM symptoms worsened when cancer recurred
in four cases, except in one case that was not described
in detail. In the two cases including our case, cancer re-
lapses were detected by exacerbation of DM symptoms,
although these were not preceded by cancer relapses in
the other two cases. Cancer recurrence occurred within
3 to 7 months after surgery in all cases, except for the
case we present here. Systemic chemotherapies were ap-
plied in all four cases, and three of the four patients died
from disease. Based on these findings, patients with
paraneoplastic disease were suggested to have resistance
to surgery, although poor prognoses compared with
common colorectal cancer could not be determined
because of the small sample size. The present case dem-
onstrated good outcomes with resection of metastatic
disease and relatively long disease-free survival.
Most patients presented with exacerbating DM symp-
toms when the colorectal cancer relapsed. It is important
to check the exacerbation of DM symptoms, including
skin erythema and dysphagia, when following patients
with paraneoplastic DM. Few studies reported recurring
cases of DM and other cancers. These reports demon-
strated the association between DM symptoms and cancer
recurrence in patients with ovarian cancer [15], breast
cancer [16], and bladder cancer [17], supporting our
Fig. 2 a 4.7 cm × 4.6 cm-sized type 2 tumor in the rectosigmoid colon. b The pathological diagnosis was moderately differented
adenocarcinoma (HE staining). c CT scan revealed a 30-mm tumor in segment 7 of the liver. d Metastatic adenocarcinoma was confirmed in the
histopathological examination
Ono et al. Surgical Case Reports  (2017) 3:3 Page 3 of 4
findings in colorectal cancer. The present report describes
a rare case with a good outcome of resecting liver metas-
tases that were detected early by exacerbation of skin
symptoms before annual examination in a patient with
DM accompanied by rectal cancer.
Conclusions
In conclusion, patients with paraneoplastic DM generally
have poor prognoses with treatment resistance. How-
ever, the present report demonstrates that there are
several cases with good, long-term results with resection
of metastatic diseases. It is important to check for the
exacerbation of DM symptoms because this symptom
sometimes appears to precede cancer relapse in the
follow-up of patients with colorectal cancer and DM.
Abbreviations
CEA: Carcinoembryonic antigen; CT: Computed tomography;
DM: Dermatomyositis
Acknowledgements
None
Authors’ contributions
All authors participated in the operation or management of the patient in
this case report. KO and TT underwent the operation. KO and MS drafted
and revised the manuscript. TT is the chairperson of our department and
supervised the writing of the manuscript. All authors read and approved the
final manuscript.
Authors’ information
All authors are surgeons of the National Hospital Organization
Higashihiroshima Medical Center.
Competing interests
The authors declare that they have no competing interests.
Consent for publication
Written informed consent was obtained from the patient for the publication
of this case report and any accompanying images. A copy of the written
consent is available for review by the Editor-in-Chief of this journal.
Author details
1Department of Surgery, National Hospital Organization Higashihiroshima
Medical Center, 513, Jike, Saijyo-cho, Higashihiroshima, Hiroshima 739-0041,
Japan. 2Department of Gastroenterological and transplant Surgery, Applied
life sciences, Institute of Biomedical & Health sciences, Hiroshima University,
Hiroshima, Japan.
Received: 15 October 2016 Accepted: 20 December 2016
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Table 1 Reported cases
Case Author (year) Sex Age Location Stage Recurrence
site
Histological
type
Disease-free
interval
DM symptom related
to the recurrence
Treatment for
recurrent disease
Outcome (time
after recurrence)
1 Ishii S (1990) F 53 Rectum I Local Mod. 4 months Present Chemotherapy DOD (2 months)
2 Yamada K
(1999)
M 58 Colon IV Liver Mod. 3 months ND Chemotherapy DOD (2 years)
3 Landriscina M
(2013)
F 71 Colon IV Lung ND 7 months Present Chemotherapy DOD (ND)
4 Nagano Y
(2015)
F 44 Rectum II Lung and
liver
Mod. 4 months Present Chemotherapy Alive with disease
(7 months)
5 Present case
(2016)
M 42 Rectum III Liver Mod. 21 months Present Resection NED (2 years)
DM dermatomyositis, Mod. moderately differentiated adenocarcinoma, ND not described, DOD died of disease, NED no evidence of disease
Ono et al. Surgical Case Reports  (2017) 3:3 Page 4 of 4


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Successful resection of liver metastasis detected by exacerbation of skin symptom in a patient with dermatomyositis accompanied by rectal cancer: a case report and literature review

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