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Annals of Clinical and Medical
Case Reports
Case Report ISSN 2639-8109 Volume 8
Ulcerative Colitis with Aseptic Abscesses Controlled by Vedolizumab: A Case Report
Fan Fan1, 2
, Li Wang1, 2
, Liming Zhang1, 2
, Yulan Liu1, 2
and Ning Chen1, 2*
1
Department of Gastroenterology, Peking University People’s Hospital, Beijing, China
2
Clinical Center of Immune-Mediated Digestive Diseases, Peking University People’s Hospital, Beijing, China
*
Corresponding author:
Ning Chen,
Department of Gastroenterology, Peking University
People’s Hospital, Beijing, China, Clinical Center
of Immune-Mediated Digestive Diseases, Peking
University People’s Hospital, Beijing, China,
Tel: (010) 88324785;
E-mail: 13683051579@139.com
Received: 12 Mar 2022
Accepted: 30 Mar 2022
Published: 05 Apr 2022
J Short Name: ACMCR
Copyright:
©2022 Ning Chen. This is an open access article distrib-
uted under the terms of the Creative Commons Attribu-
tion License, which permits unrestricted use, distribu-
tion, and build upon your work non-commercially.
Citation:
Ning Chen, Ulcerative Colitis with Aseptic Abscesses
Controlled by Vedolizumab: A Case Report. Ann Clin
Med Case Rep. 2022; V8(17): 1-4
http://www.acmcasereport.com/ 1
Keywords:
Inflammatory bowel disease; Extraintestinal
manifestations; Aseptic abscesses; Vedolizumab
1. Abstract
1.1. Introduction: Aseptic abscesses (AAs) are neutrophilic in-
filtrative lesions that often coincide with systemic inflammatory
disorders such as inflammatory bowel diseases (IBD). According
to recent literature, medical therapies in IBD with AAs include
corticosteroid, immunosuppressants and anti-TNFα biologics.
1.2. Report: Herein we reported a patient of ulcerative colitis with
AAs and other extraintestinal manifestations treated with vedoli-
zumab that showed a very inspiring outcome.
1.3. Discussion: It’s the first successful performance of vedoli-
zumab treating moderate-severe ulcerative colitis with aseptic ab-
scesses. This case may provide more therapeutic alternatives for
patients with similar conditions in the future and offer hints for
future study.
2. Introduction
Aseptic abscesses [AAs] are focal lesions that characterized as
inflammatory neutrophils infiltration, negative blood and aspirate
pathogen culture, refractory to broad spectrum antibiotics and sen-
sitive to immunosuppressive therapies [1].Since first described 30
years ago by Andre et al, dozens of cases of aseptic abscesses have
been found to associate with Inflammatory bowel diseases [IBD]
[2]. AAs are currently accepted as one of the extraintestinal man-
ifestations [EIMs] of IBD, although much rarely presented com-
pared to other types of EIMs involving musculoskeletal systems,
skin, eye and hepatobiliary tract [3]. Previous reports showed AA
in IBD was successfully treated with corticosteroid, azathioprine,
cyclophosphamide, methotrexate, infliximab, adalimumab and
surgical modalities [4]. In recent years, a novel monoclonal bio-
logics vedolizumab, was developed to treat IBD which, by target-
ing at the α4β7 integrin, selectively blocks lymphocytes trafficking
to the gastrointestinal tract [5]. Efficacy of vedolizumab to extrain-
testinal manifestations of IBD is still inconclusive, particularly its
efficacy to aseptic abscesses. Herein, we report a case of ulcerative
colitis with aseptic abscesses and musculoskeletal manifestations
successfully treated with vedolizumab.
3. Case Report
A 33-year-old woman presented with history of ulcerative colitis
for 4 years. Mesalazine was effective at the beginning 4 years ago,
but gradually became inadequate. Corticosteroid was administered
and showed promising effect during induction. However, bloody
stool recurred during tapering. By the time she presented, she had
fever with body temperature as 38.9℃, abdominal pain and bloody
stool 4-6 times per day. She also complained multiple peripheral
arthralgia affecting hips, knees, ankles, heels and interphalangeal
joints. Physical exam showed tender in left side abdomen. Labo-
ratory tests showed peripheral white cell count 14.6×109
/L, hemo-
globin 97g/L, C-reactive protein 80.6mg/L, erythrocyte sedimen-
tation rate 70mm/h, albumin 33.6g/L. Liver and renal function and
electrolyte tests were unremarkable. We conducted comprehensive
infectious pathogen screening tests including blood culture for
bacteria and fungi, fungal 1,3-β-D-glucan and galactomannan test,
test for Clostridium difficile toxin, microscopic pathogen detec-
tion for stool smear, T-cell spot test for tuberculosis, nucleic acid
test for CMV and EBV that were all negative. A contrast-enhanced
CT scan was performed which showed diffused thickening of left-
http://www.acmcasereport.com/ 2
Volume 8 Issue 17 -2022 Case Report
side colon wall, multiple low enhanced lesions in liver and kidneys
and multiple newly appearing lymphadenopathies in mediastinal,
mesenteric and para-abdominal aortic areas with some particularly
enlarged abdominal lymph nodes that were up to 2-3cm diameters
with central liquefactive necrosis [Figure 2B]. Those image results
were confirmed by enhanced MR scan [Figure 2A]. Colonoscopy
demonstrated active ulcerative colitis that affected from the trans-
verse colon near the hepatic flexure to the descending colon [Fig-
ure 1A, 1B]. She was diagnosed as ulcerative colitis. Pathological
nature of the enlarged abdominal lymphadenopathies and hepatic
and renal lesions were considered including multi-organ infectious
lesions, which is reasonable considering her immunocompromised
status due to colitis and past history of using corticosteroid, and
lymphoma which have been reported repeatedly in patients of in-
flammatory bowel disease with lymphadenopathy. She was admin-
istrated with broad spectrum antibiotics for 3 weeks (Ertapenem 1g
every day for a week, Cefoperazone/Sulbactam 3g every 8 hours
with Ornidazole 0.5g every 12 hours for a week and levofloxacin
0.4g every day with Ornidazole 0.5g every 12 hours for a week).
Her peripheral white cell count was moderately improved. How-
ever, activity of bowel disease and other manifestations showed
much less alleviation. PET-CT scan was conducted that showed no
indication of lymphoma. We also performed endoscopic ultraso-
nography with fine needle aspiration [EUS-FNA]. Results of EUS
coincided with CT and MR scan and FNA acquired 2ml of puru-
lent fluid which was further sent to bacteria, fungi and tuberculo-
sis cultures, Next Generation Sequencing [NGS] for pathogens,
pathological examination and cytological smears [Figure 3]. Evi-
dence of infectious pathogen was negative and pathological exam
demonstrated necrotic tissue with neutrophils infiltration. Hip
joints MR scan was also performed with experienced rheumatolo-
gist’s evaluation that confirmed polyarthritis. We finally diagnosed
her as ulcerative colitis (chronic relapse, pancolitis, moderate to
severe activity), aseptic abscesses (mesenteric lymph nodes, liver
and kidneys), musculoskeletal manifestation (peripheral arthrop-
athy) and recommended vedolizumab for her. We then witnessed
a dramatic remission of her intestinal and articular symptoms and
improvement of her lymphatic enlargement, hepatic and renal le-
sions that were confirmed by colonoscopy and MR scan at the time
of 4-month follow-up [Figure 1C, 1D, 2C and 2D]. Her hemoglo-
bin and albumin levels gradually recovered to normal and inflam-
matory markers such as CRP decreased accordingly. Till the time
this report written, it has been 9 months since vedolizumab therapy
began, she came back to hospital periodically for continuous treat-
ment and surveillance, and there’s no sign of any sort of relapse.
Figure 1: Colonoscopy images of transverse colon and descending colon before and after 4 months of treatment.
Colonoscopy showed friable mucosa, diffused mucosal congestion and edema with flaky ulcers and erosions covered with mucopurulent secretions and
lack of vascular pattern in the descending colon (A) and transverse colon (B) at the time of admission and recovered mucosa with nascent granulation
tissue and inflammatory polyps in the descending colon (C) and transverse colon (D) 4 months after treated with vedolizumab.
http://www.acmcasereport.com/ 3
Volume 8 Issue 17 -2022 Case Report
Figure 2:Abdominal radiological images demonstrating hepatic and renal lesions and aseptic abscesses before and after 4 months of treatment.
MRI (A, C) and CT (B, D) scan of upper-abdomen showed multiple lesions in live and kidneys (green arrows) and enlarged abdominal lymph node
with central liquefactive necrosis (yellow arrows) at the time of admission (A, B). After treated with vedolizumab, the lesions decreased in sizes (B, D).
Figure 3: Endoscopic ultrasonography and fine needle aspiration (EUS-FNA) of the enlarged and necrotic lymph nodes.
EUS demonstrated abdominal enlarged lymph nodes with central liquefactive necrosis (A). Fine needle aspiration was performed and 2ml of purulent
fluid was acquired (B and C).
4. Discussion
To our knowledge, this is the first case of ulcerative colitis with
aseptic abscesses treated with vedolizumab that showed a prom-
ising result during at least 9 months follow-up. Aseptic abscesses
were first described in 1995 by Andre et al. in patients with ab-
scesses that could not be explained by infection because of un-
responsiveness to antibiotics and sensitivity to corticosteroid [2].
It often associates with systemic inflammatory conditions, mostly
IBD as one of extraintestinal manifestation [6, 7]. Similar to neu-
trophilic dermatosis such as Pyoderma Gangrenosum and Sweet’s
Syndrome which are two types of more often seen dermatological
manifestations of IBD, histopathological characteristics of AA is
sterile predominant neutrophils infiltration of the deep tissues [6].
There is currently no diagnostic criterion for AA. However, An-
dre and colleagues came up with a set of common characteristics
base on case series for clinicians to evaluate AA: (1) deep abscess-
es with neutrophilic features; (2) negative findings of causative
infectious pathogen from serological tests and cultures of blood
and aspiration sample; (3) failure of broad-spectrum antibiotics;
(4) clinical improvement on corticosteroid therapy with or without
additional immunosuppressant which is confirmed by radiologic
evidence [3]. According to the latest literature review by Yama-
guchi. Y and teammates, 43 patients of IBD-associated AA were
identified in MEDLINE from 1994 to 2020. Patients may present
with fever, abdominal pain, weight loss and diarrhea. Lesions may
be located at spleen, liver, lymph nodes, muscles, kidneys, sternum
and other locations in very rare cases. Patients may coincide with
other EIM of IBD such as arthritis, myalgia, neutrophilic dermato-
sis, aphthous ulcer, panniculitis and polyneuropathy. Several treat-
ments had been reported including corticosteroids which is most
often mentioned, cyclophosphamide, azathioprine, methotrexate,
granulocytapheresis, adalimumab, infliximab and surgical proce-
dures such as splenectomy, incision and drainage, laparoscopic
biopsy of lymph nodes [1]. In this case, we regarded this patient
as IBD-associated AAs according to results of pathogen tests, his-
topathological findings and therapeutic effect of broad-spectrum
antibiotics. However, other differential diagnoses such as oppor-
tunistic infection, lymphoma and tuberculosis infection couldn’t
http://www.acmcasereport.com/ 4
Volume 8 Issue 17 -2022 Case Report
be excluded. Thus, among several therapeutic options including
corticosteroid, anti-TNF biologics, and anti-Integrin biologics,
we deemed vedolizumab as the optimal choice for her from the
perspective of safety because of its intestinal selectivity. Outcome
of this treatment was inspiring for not only remission of colonic
mucosal inflammation but also alleviation of AAs and arthralgias.
Inflammatory bowel disease is not limited to the digestive tract
and is regarded as a systemic immune disorder with nearly one
half of patients experiencing extraintestinal manifestations which
involve multiple organs and systems including musculoskele-
tal system, skin, ocular organ and hepatobiliary tract and others
[8]. The pathophysiological mechanism of EIMs is unclear with
several hypotheses. Either they could be the result of intestinal
immune activation with inflammatory cells and cytokines subse-
quently trafficking or diffusing to extraintestinal target organs, or
they, together with gastrointestinal tract, could be simultaneously
involved by systemic immune disturbance [3]. Vedolizumab is a
gut-selective antibody to α4β7 integrin for treatment of moder-
ate-severe IBD. Studies have shown long-term effectiveness and
a favorable safety profile of vedolizumab with low incidence rates
of serious infections and malignancies at least partially due to its
selectivity [9, 10]. It is uncertain whether a gut-selective action is
suitable for controlling EIMs such as aseptic abscesses and mus-
culoskeletal manifestations. In a systemic review worked by Han-
zel. J and team, vedolizumab may be effective for musculoskeletal
EIMs associated with disease activity [3]. There were case reports
suggesting that vedolizumab could be effective for pyoderma gan-
grenosum according to Groudan K. et al., Fleisher M. et al. and
Shibuya T. et al. [11, 12, 13]. However, there were no randomized
controlled trials that specifically study the efficacy of vedolizumab
for IBD related EIMs especially aseptic abscesses and arthropathy,
and evidence from different observational studies are of modest
quality. Thus, large-scale well-designed prospective studies are
needed in the future in order to definitively answer such questions.
References
1. Yamaguchi Y, Nakagawa M, Nakagawa S. Rapidly Progressing
Aseptic Abscesses in a Patient with Ulcerative Colitis. Intern Med.
2021; 60(5): 725-730.
2. Sakharpe AK, Sakharpe AK, Mirmanesh M. A case and review of
aseptic liver abscess in Crohn’s disease. Int J Colorectal Dis. 2016;
31(3): 787-788.
3. Hanzel J, Ma C, Casteele NV, Khanna R, Jairath V, Feagan BG.
Vedolizumab and Extraintestinal Manifestations in Inflammatory
Bowel Disease [published correction appears in Drugs. 2021 Aug
28;:]. Drugs. 2021; 81(3): 333-347.
4. Bollegala N, Khan R, Scaffidi MA. Aseptic Abscesses and Inflam-
matory Bowel Disease: Two Cases and Review of Literature. Can J
Gastroenterol Hepatol. 2017; 2017: 5124354.
5. Feagan BG, Sandborn WJ, Colombel JF. Incidence of Arthritis/Ar-
thralgia in Inflammatory Bowel Disease with Long-term Vedolizum-
ab Treatment: Post Hoc Analyses of the GEMINI Trials. J Crohns
Colitis. 2019; 13(1): 50-57.
6. Fillman H, Riquelme P, Sullivan PD, Mansoor AM. Aseptic abscess
syndrome. BMJ Case Rep. 2020; 13(10): e236437.
7. Antonelli E, Bassotti G, Tramontana M. Dermatological Manifes-
tations in Inflammatory Bowel Diseases. J Clin Med. 2021; 10(2):
364.
8. Vavricka SR, Schoepfer A, Scharl M, Lakatos PL, Navarini A,
Rogler G. Extraintestinal Manifestations of Inflammatory Bowel
Disease. Inflamm Bowel Dis. 2015; 21(8): 1982-1992.
9. Loftus EV Jr, Colombel JF, Feagan BG. Long-term Efficacy of
Vedolizumab for Ulcerative Colitis. J Crohns Colitis. 2017; 11(4):
400-411.
10. Colombel JF, Sands BE, Rutgeerts P. The safety of vedolizumab for
ulcerative colitis and Crohn’s disease. Gut. 2017; 66(5): 839-851.
11. Shibuya T, Haga K, Saeki M. Pyoderma gangrenosum in an ulcer-
ative colitis patient during treatment with vedolizumab responded
favorably to adsorptive granulocyte and monocyte apheresis. J Clin
Apher. 2020; 35(5): 488-492.
12. Groudan K, Gupta K, Singhania R. Vedolizumab (Entyvio®) for the
Treatment of Pyoderma Gangrenosum in a Crohn’s Disease Patient.
Cureus. 2021; 13(1): e12582.
13. Fleisher M, Marsal J, Lee SD. Effects of Vedolizumab Therapy on
Extraintestinal Manifestations in Inflammatory Bowel Disease. Dig
Dis Sci. 2018; 63(4): 825-833.

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Ulcerative colitis with aseptic abscesses controlled by Vedolizumab: A case report

  • 1. Annals of Clinical and Medical Case Reports Case Report ISSN 2639-8109 Volume 8 Ulcerative Colitis with Aseptic Abscesses Controlled by Vedolizumab: A Case Report Fan Fan1, 2 , Li Wang1, 2 , Liming Zhang1, 2 , Yulan Liu1, 2 and Ning Chen1, 2* 1 Department of Gastroenterology, Peking University People’s Hospital, Beijing, China 2 Clinical Center of Immune-Mediated Digestive Diseases, Peking University People’s Hospital, Beijing, China * Corresponding author: Ning Chen, Department of Gastroenterology, Peking University People’s Hospital, Beijing, China, Clinical Center of Immune-Mediated Digestive Diseases, Peking University People’s Hospital, Beijing, China, Tel: (010) 88324785; E-mail: 13683051579@139.com Received: 12 Mar 2022 Accepted: 30 Mar 2022 Published: 05 Apr 2022 J Short Name: ACMCR Copyright: ©2022 Ning Chen. This is an open access article distrib- uted under the terms of the Creative Commons Attribu- tion License, which permits unrestricted use, distribu- tion, and build upon your work non-commercially. Citation: Ning Chen, Ulcerative Colitis with Aseptic Abscesses Controlled by Vedolizumab: A Case Report. Ann Clin Med Case Rep. 2022; V8(17): 1-4 http://www.acmcasereport.com/ 1 Keywords: Inflammatory bowel disease; Extraintestinal manifestations; Aseptic abscesses; Vedolizumab 1. Abstract 1.1. Introduction: Aseptic abscesses (AAs) are neutrophilic in- filtrative lesions that often coincide with systemic inflammatory disorders such as inflammatory bowel diseases (IBD). According to recent literature, medical therapies in IBD with AAs include corticosteroid, immunosuppressants and anti-TNFα biologics. 1.2. Report: Herein we reported a patient of ulcerative colitis with AAs and other extraintestinal manifestations treated with vedoli- zumab that showed a very inspiring outcome. 1.3. Discussion: It’s the first successful performance of vedoli- zumab treating moderate-severe ulcerative colitis with aseptic ab- scesses. This case may provide more therapeutic alternatives for patients with similar conditions in the future and offer hints for future study. 2. Introduction Aseptic abscesses [AAs] are focal lesions that characterized as inflammatory neutrophils infiltration, negative blood and aspirate pathogen culture, refractory to broad spectrum antibiotics and sen- sitive to immunosuppressive therapies [1].Since first described 30 years ago by Andre et al, dozens of cases of aseptic abscesses have been found to associate with Inflammatory bowel diseases [IBD] [2]. AAs are currently accepted as one of the extraintestinal man- ifestations [EIMs] of IBD, although much rarely presented com- pared to other types of EIMs involving musculoskeletal systems, skin, eye and hepatobiliary tract [3]. Previous reports showed AA in IBD was successfully treated with corticosteroid, azathioprine, cyclophosphamide, methotrexate, infliximab, adalimumab and surgical modalities [4]. In recent years, a novel monoclonal bio- logics vedolizumab, was developed to treat IBD which, by target- ing at the α4β7 integrin, selectively blocks lymphocytes trafficking to the gastrointestinal tract [5]. Efficacy of vedolizumab to extrain- testinal manifestations of IBD is still inconclusive, particularly its efficacy to aseptic abscesses. Herein, we report a case of ulcerative colitis with aseptic abscesses and musculoskeletal manifestations successfully treated with vedolizumab. 3. Case Report A 33-year-old woman presented with history of ulcerative colitis for 4 years. Mesalazine was effective at the beginning 4 years ago, but gradually became inadequate. Corticosteroid was administered and showed promising effect during induction. However, bloody stool recurred during tapering. By the time she presented, she had fever with body temperature as 38.9℃, abdominal pain and bloody stool 4-6 times per day. She also complained multiple peripheral arthralgia affecting hips, knees, ankles, heels and interphalangeal joints. Physical exam showed tender in left side abdomen. Labo- ratory tests showed peripheral white cell count 14.6×109 /L, hemo- globin 97g/L, C-reactive protein 80.6mg/L, erythrocyte sedimen- tation rate 70mm/h, albumin 33.6g/L. Liver and renal function and electrolyte tests were unremarkable. We conducted comprehensive infectious pathogen screening tests including blood culture for bacteria and fungi, fungal 1,3-β-D-glucan and galactomannan test, test for Clostridium difficile toxin, microscopic pathogen detec- tion for stool smear, T-cell spot test for tuberculosis, nucleic acid test for CMV and EBV that were all negative. A contrast-enhanced CT scan was performed which showed diffused thickening of left-
  • 2. http://www.acmcasereport.com/ 2 Volume 8 Issue 17 -2022 Case Report side colon wall, multiple low enhanced lesions in liver and kidneys and multiple newly appearing lymphadenopathies in mediastinal, mesenteric and para-abdominal aortic areas with some particularly enlarged abdominal lymph nodes that were up to 2-3cm diameters with central liquefactive necrosis [Figure 2B]. Those image results were confirmed by enhanced MR scan [Figure 2A]. Colonoscopy demonstrated active ulcerative colitis that affected from the trans- verse colon near the hepatic flexure to the descending colon [Fig- ure 1A, 1B]. She was diagnosed as ulcerative colitis. Pathological nature of the enlarged abdominal lymphadenopathies and hepatic and renal lesions were considered including multi-organ infectious lesions, which is reasonable considering her immunocompromised status due to colitis and past history of using corticosteroid, and lymphoma which have been reported repeatedly in patients of in- flammatory bowel disease with lymphadenopathy. She was admin- istrated with broad spectrum antibiotics for 3 weeks (Ertapenem 1g every day for a week, Cefoperazone/Sulbactam 3g every 8 hours with Ornidazole 0.5g every 12 hours for a week and levofloxacin 0.4g every day with Ornidazole 0.5g every 12 hours for a week). Her peripheral white cell count was moderately improved. How- ever, activity of bowel disease and other manifestations showed much less alleviation. PET-CT scan was conducted that showed no indication of lymphoma. We also performed endoscopic ultraso- nography with fine needle aspiration [EUS-FNA]. Results of EUS coincided with CT and MR scan and FNA acquired 2ml of puru- lent fluid which was further sent to bacteria, fungi and tuberculo- sis cultures, Next Generation Sequencing [NGS] for pathogens, pathological examination and cytological smears [Figure 3]. Evi- dence of infectious pathogen was negative and pathological exam demonstrated necrotic tissue with neutrophils infiltration. Hip joints MR scan was also performed with experienced rheumatolo- gist’s evaluation that confirmed polyarthritis. We finally diagnosed her as ulcerative colitis (chronic relapse, pancolitis, moderate to severe activity), aseptic abscesses (mesenteric lymph nodes, liver and kidneys), musculoskeletal manifestation (peripheral arthrop- athy) and recommended vedolizumab for her. We then witnessed a dramatic remission of her intestinal and articular symptoms and improvement of her lymphatic enlargement, hepatic and renal le- sions that were confirmed by colonoscopy and MR scan at the time of 4-month follow-up [Figure 1C, 1D, 2C and 2D]. Her hemoglo- bin and albumin levels gradually recovered to normal and inflam- matory markers such as CRP decreased accordingly. Till the time this report written, it has been 9 months since vedolizumab therapy began, she came back to hospital periodically for continuous treat- ment and surveillance, and there’s no sign of any sort of relapse. Figure 1: Colonoscopy images of transverse colon and descending colon before and after 4 months of treatment. Colonoscopy showed friable mucosa, diffused mucosal congestion and edema with flaky ulcers and erosions covered with mucopurulent secretions and lack of vascular pattern in the descending colon (A) and transverse colon (B) at the time of admission and recovered mucosa with nascent granulation tissue and inflammatory polyps in the descending colon (C) and transverse colon (D) 4 months after treated with vedolizumab.
  • 3. http://www.acmcasereport.com/ 3 Volume 8 Issue 17 -2022 Case Report Figure 2:Abdominal radiological images demonstrating hepatic and renal lesions and aseptic abscesses before and after 4 months of treatment. MRI (A, C) and CT (B, D) scan of upper-abdomen showed multiple lesions in live and kidneys (green arrows) and enlarged abdominal lymph node with central liquefactive necrosis (yellow arrows) at the time of admission (A, B). After treated with vedolizumab, the lesions decreased in sizes (B, D). Figure 3: Endoscopic ultrasonography and fine needle aspiration (EUS-FNA) of the enlarged and necrotic lymph nodes. EUS demonstrated abdominal enlarged lymph nodes with central liquefactive necrosis (A). Fine needle aspiration was performed and 2ml of purulent fluid was acquired (B and C). 4. Discussion To our knowledge, this is the first case of ulcerative colitis with aseptic abscesses treated with vedolizumab that showed a prom- ising result during at least 9 months follow-up. Aseptic abscesses were first described in 1995 by Andre et al. in patients with ab- scesses that could not be explained by infection because of un- responsiveness to antibiotics and sensitivity to corticosteroid [2]. It often associates with systemic inflammatory conditions, mostly IBD as one of extraintestinal manifestation [6, 7]. Similar to neu- trophilic dermatosis such as Pyoderma Gangrenosum and Sweet’s Syndrome which are two types of more often seen dermatological manifestations of IBD, histopathological characteristics of AA is sterile predominant neutrophils infiltration of the deep tissues [6]. There is currently no diagnostic criterion for AA. However, An- dre and colleagues came up with a set of common characteristics base on case series for clinicians to evaluate AA: (1) deep abscess- es with neutrophilic features; (2) negative findings of causative infectious pathogen from serological tests and cultures of blood and aspiration sample; (3) failure of broad-spectrum antibiotics; (4) clinical improvement on corticosteroid therapy with or without additional immunosuppressant which is confirmed by radiologic evidence [3]. According to the latest literature review by Yama- guchi. Y and teammates, 43 patients of IBD-associated AA were identified in MEDLINE from 1994 to 2020. Patients may present with fever, abdominal pain, weight loss and diarrhea. Lesions may be located at spleen, liver, lymph nodes, muscles, kidneys, sternum and other locations in very rare cases. Patients may coincide with other EIM of IBD such as arthritis, myalgia, neutrophilic dermato- sis, aphthous ulcer, panniculitis and polyneuropathy. Several treat- ments had been reported including corticosteroids which is most often mentioned, cyclophosphamide, azathioprine, methotrexate, granulocytapheresis, adalimumab, infliximab and surgical proce- dures such as splenectomy, incision and drainage, laparoscopic biopsy of lymph nodes [1]. In this case, we regarded this patient as IBD-associated AAs according to results of pathogen tests, his- topathological findings and therapeutic effect of broad-spectrum antibiotics. However, other differential diagnoses such as oppor- tunistic infection, lymphoma and tuberculosis infection couldn’t
  • 4. http://www.acmcasereport.com/ 4 Volume 8 Issue 17 -2022 Case Report be excluded. Thus, among several therapeutic options including corticosteroid, anti-TNF biologics, and anti-Integrin biologics, we deemed vedolizumab as the optimal choice for her from the perspective of safety because of its intestinal selectivity. Outcome of this treatment was inspiring for not only remission of colonic mucosal inflammation but also alleviation of AAs and arthralgias. Inflammatory bowel disease is not limited to the digestive tract and is regarded as a systemic immune disorder with nearly one half of patients experiencing extraintestinal manifestations which involve multiple organs and systems including musculoskele- tal system, skin, ocular organ and hepatobiliary tract and others [8]. The pathophysiological mechanism of EIMs is unclear with several hypotheses. Either they could be the result of intestinal immune activation with inflammatory cells and cytokines subse- quently trafficking or diffusing to extraintestinal target organs, or they, together with gastrointestinal tract, could be simultaneously involved by systemic immune disturbance [3]. Vedolizumab is a gut-selective antibody to α4β7 integrin for treatment of moder- ate-severe IBD. Studies have shown long-term effectiveness and a favorable safety profile of vedolizumab with low incidence rates of serious infections and malignancies at least partially due to its selectivity [9, 10]. It is uncertain whether a gut-selective action is suitable for controlling EIMs such as aseptic abscesses and mus- culoskeletal manifestations. In a systemic review worked by Han- zel. J and team, vedolizumab may be effective for musculoskeletal EIMs associated with disease activity [3]. There were case reports suggesting that vedolizumab could be effective for pyoderma gan- grenosum according to Groudan K. et al., Fleisher M. et al. and Shibuya T. et al. [11, 12, 13]. However, there were no randomized controlled trials that specifically study the efficacy of vedolizumab for IBD related EIMs especially aseptic abscesses and arthropathy, and evidence from different observational studies are of modest quality. Thus, large-scale well-designed prospective studies are needed in the future in order to definitively answer such questions. References 1. Yamaguchi Y, Nakagawa M, Nakagawa S. Rapidly Progressing Aseptic Abscesses in a Patient with Ulcerative Colitis. Intern Med. 2021; 60(5): 725-730. 2. Sakharpe AK, Sakharpe AK, Mirmanesh M. A case and review of aseptic liver abscess in Crohn’s disease. Int J Colorectal Dis. 2016; 31(3): 787-788. 3. Hanzel J, Ma C, Casteele NV, Khanna R, Jairath V, Feagan BG. Vedolizumab and Extraintestinal Manifestations in Inflammatory Bowel Disease [published correction appears in Drugs. 2021 Aug 28;:]. Drugs. 2021; 81(3): 333-347. 4. Bollegala N, Khan R, Scaffidi MA. Aseptic Abscesses and Inflam- matory Bowel Disease: Two Cases and Review of Literature. Can J Gastroenterol Hepatol. 2017; 2017: 5124354. 5. Feagan BG, Sandborn WJ, Colombel JF. Incidence of Arthritis/Ar- thralgia in Inflammatory Bowel Disease with Long-term Vedolizum- ab Treatment: Post Hoc Analyses of the GEMINI Trials. J Crohns Colitis. 2019; 13(1): 50-57. 6. Fillman H, Riquelme P, Sullivan PD, Mansoor AM. Aseptic abscess syndrome. BMJ Case Rep. 2020; 13(10): e236437. 7. Antonelli E, Bassotti G, Tramontana M. Dermatological Manifes- tations in Inflammatory Bowel Diseases. J Clin Med. 2021; 10(2): 364. 8. Vavricka SR, Schoepfer A, Scharl M, Lakatos PL, Navarini A, Rogler G. Extraintestinal Manifestations of Inflammatory Bowel Disease. Inflamm Bowel Dis. 2015; 21(8): 1982-1992. 9. Loftus EV Jr, Colombel JF, Feagan BG. Long-term Efficacy of Vedolizumab for Ulcerative Colitis. J Crohns Colitis. 2017; 11(4): 400-411. 10. Colombel JF, Sands BE, Rutgeerts P. The safety of vedolizumab for ulcerative colitis and Crohn’s disease. Gut. 2017; 66(5): 839-851. 11. Shibuya T, Haga K, Saeki M. Pyoderma gangrenosum in an ulcer- ative colitis patient during treatment with vedolizumab responded favorably to adsorptive granulocyte and monocyte apheresis. J Clin Apher. 2020; 35(5): 488-492. 12. Groudan K, Gupta K, Singhania R. Vedolizumab (Entyvio®) for the Treatment of Pyoderma Gangrenosum in a Crohn’s Disease Patient. Cureus. 2021; 13(1): e12582. 13. Fleisher M, Marsal J, Lee SD. Effects of Vedolizumab Therapy on Extraintestinal Manifestations in Inflammatory Bowel Disease. Dig Dis Sci. 2018; 63(4): 825-833.