CASE REPORT Open Access

A case of flank pain caused by ureteralintussusception accompanied with ureteralpolypYang Dong†, Wen-da Zhang†, Tao Fan†, Lin Hao, Jia-he Zhou, Wei-ming Ma and Cong-hui Han*

Abstract

Background: Ureteral intussusception, a rarely reported unique condition, occurs primarily as a complication ofureteric tumours.

Case presentation: We present a case of ureteral intussusception accompanied with a large ureteral polypperiodically protruding into the bladder cavity occurring in a 56-year-old man who experienced vague flank painand intermittent haematuria. The patient was successfully treated by ureteroscopic cauterization combined withpartial ureterectomy with reanastomosis.

Conclusions: This is the first report that describes polyp-related ureteral intussusception using comprehensive andrepresentative ureteroscopic images and video. Our findings suggest that ureteroscopy is vital for diagnosis.Extensive biopsies through ureteroscopy are less invasive, and make it easier to exclude the presence of ureteralmalignancies. Ureteroscopic resection of the whole polyp with its stalk and intussusceptum using Holmium: YAGlaser did not seem viable in this case. However, cauterization of partial polyp tissues followed by open surgery forsegmental resection of the ureter with reanastomosis is helpful in controlling such patient well-being.

Keywords: Ureteral intussusception, Ureteral polyp, Ureteroscopy

BackgroundUreteral intussusception is a rarely reported unique con-dition, in which the proximal ureteral wall telescopesinto the distal lumen. It often develops slowly and oc-curs secondary to ureteral neoplasms; however, it is oc-casionally caused by calculi or endoscopic surgicalprocedures. Due to the lack of awareness regarding thiscondition, ureteral intussusception is often unsuspectedand misdiagnosed. Here, we present a case of ureteralintussusception accompanied with ureteral polyp and, toour knowledge, this is the first report that provides com-prehensive and representative ureteroscopic images andvideo depicting the case.

Case presentationA 56-year-old man presented with a 1-week history ofvague pain in the right flank and suprapubic region, andintermittent haematuria. He recalled having similar mildattacks periodically 2 years before that remitted withouttreatment. He denied any significant medical history, thehabit of smoking and explosion to any solvents or che-micals. Bladder neoplasm was initially suspected by out-patient colour Doppler ultrasonography and cystoscopyat a local hospital 3 days prior to admission. No abnor-malities were observed upon physical examination, sero-logical examination, and urine cytology after admission.Computed tomography (CT) revealed an enlargement inthe inferior part of the right ureter with a suspectedsolid mass (Fig. 1a). Further intravenous urogram (IVU)revealed a “sponge-like” filling defect in the right lowerenlarged ureteral lumen and right ureteral orifice inside

© The Author(s). 2020 Open Access This article is licensed under a Creative Commons Attribution 4.0 International License,which permits use, sharing, adaptation, distribution and reproduction in any medium or format, as long as you giveappropriate credit to the original author(s) and the source, provide a link to the Creative Commons licence, and indicate ifchanges were made. The images or other third party material in this article are included in the article's Creative Commonslicence, unless indicated otherwise in a credit line to the material. If material is not included in the article's Creative Commonslicence and your intended use is not permitted by statutory regulation or exceeds the permitted use, you will need to obtainpermission directly from the copyright holder. To view a copy of this licence, visit http://creativecommons.org/licenses/by/4.0/.The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/) applies to thedata made available in this article, unless otherwise stated in a credit line to the data.

* Correspondence: 201706014207bjk@ncist.edu.cn†Yang Dong, Wen-da Zhang and Tao Fan contributed equally to this work.Department of Urology, Xuzhou Central Hospital Affiliated to XuzhouMedical University, Jiefang South Road, No. 199, Xuzhou, Jiangsu, China

Dong et al. BMC Nephrology (2020) 21:246 https://doi.org/10.1186/s12882-020-01904-8

the bladder, but no dilation of the upper urinary tractand renal pelvis (Fig. 1b). Ureteral tumour was detectedpreoperatively.Ureteroscopy revealed that the right lower ureteral

lumen was occupied by multiple white-grey polypoid tu-mours, floating in the ureter that periodically moved inand out of the bladder through the ureteral orifice. Bi-opsy demonstrated an inflammatory polyp. This polyptogether with terminal lobulations was approximately

7.0-cm in length containing a large pedicle that origi-nated from the tip of the proximal ureter. The ureterallumen terminated at the base of the polyp, but a ureteralorifice was observed in the centre of the proximal ped-icle (Fig. 2a). After exploring the interior of the pedicleby ureteroscopy (Fig. 2b), we confirmed that the upperpartial pedicle was an intussuscepted segment of the ur-eter approximately 2.5-cm in length and averaging 8.0-mm in diameter (supplementary video). The patient was

Fig. 1 Computed tomography and intravenous urogram findings (a) Computed tomography shows an enlargement in the inferior part of theright ureter with a suspected solid mass; (b) Intravenous urogram shows a “sponge-like” filling defect in the right distal dilated ureteral lumen(black arrow) without dilation of the upper urinary tract and renal pelvis (white arrow), and an oval-shaped filling defect of the ureteral orificeinside the bladder

Fig. 2 Ureteroscopic view. Ureteroscopy showing an intussuscepted ureter protruding into the lumen of the adjacent segment (a); A wire waspassed to the renal pelvis through the lumen of the intussusceptum (b)

Dong et al. BMC Nephrology (2020) 21:246 Page 2 of 9

treated by ureteroscopic cauterization of the partialpolyp at the slimmest point of the pedicle followed byopen surgery exploration. A fusiform was found in thelower dilated thick-walled ureter and palpation revealeda firm and mobile tumour. Finally, right ureteral partialresection with reanastomosis was performed to containthe pedicle stalk and ureteral segment. Postoperative re-covery was uneventful, and pathological examinationconfirmed a fibroepithelial polyp. The double-J stent wasremoved 6 weeks after the operation. A CT scan of theabdomen performed 3months post-surgery indicatedneither uronephrosis nor any signs of polyp recurrence.

Discussion and conclusionsTo identify relevant studies up to May 2020, thePubMed database was searched using the terms (“ur-eteral” [All Fields] OR “ureter” [All Fields] OR “ureter”[MeSH Terms]) AND (“intussusception” [All Fields] OR“intussusception” [MeSH Terms]). We retrieved 101studies, of which 28 (30 cases) that published from 1937to 2019 were finally selected after review (Table 1). Ofthese, 25 cases were related to ureteral neoplasms, in-cluding 15 secondary to polyps, and 10 secondary to ma-lignant tumours. The remaining five cases included oneureteral calculus-related case, and four cases of iatro-genic complication occurring in surgical procedures.Ureteral intussusception does not occur spontan-

eously, largely because of the small ratio between ur-eteral wall thickness and lumen calibre and the limitedrange of mobility of the ureter itself, which prevents ur-eter invagination [25]. However, when a slow-growingobject, typically a benign tumour, occupies the ureter, itdraws down the proximal ureter, enters the distal dilatedureter by peristaltic activity, urine flow, and gravitationalforce, and sporadically leads to ureteral intussusception[6, 8, 19] (Fig. 3). A review of previously reported casesrevealed that intussusception is usually antegrade andmore prevalent in men on the right side. The commonmanifestations in patients include a history of intermit-tent haematuria and repeated flank pain [25, 26], butmalignant tumour-related ureteral intussusception isoften asymptomatic and occurs uniformly in patientsaged ≥50 years [25]. Some scholars proposed the poten-tial risk of ureteral ischemia because of the possible defi-ciency of the blood supply to the intussusceptum [28].However, no evidence of regional ureteral ischemic ne-crosis associated with ureteral intussusception has beenreported previously, which is compatible with the char-acteristic sluggish changes underlying intussusception.Ureteral intussusception is characterized by distinct

features, especially on IVU and contrast CT images thathave great diagnostic value. In primary lesions, ureteralintussusception often presents a “bell-shaped [5, 21,28]”, “tongue-like [24] “or “sponge-like [23] “filling

defect in an enlarged ureteral segment, with or withouthydronephrosis. The intussuscepted segment filled withcontrast material appears as a “line” sign [22], and theinvaginated ureteral lumen appears as a “V-shaped” sign[28], also described as “claw of crab -shaped” sign in theupstream from the mass [8]. Non-contrast CT scan ishelpful for revealing calculus and solid ureteral massesbut can hardly detect ureteral intussusception. Incontrast-enhanced CT images, contrast material opaci-fies both intussusceptum and distal intussuscipiens,forming a “concentric sign” [9, 22, 23, 27] or “bull’s-eyesign” [25] on axial imaging, and a “stalk-of-corn” appear-ance on coronal and sagittal imaging [25]. Ureteroscopyand intraoperative biopsy enable definitive diagnosis andare capable of distinguishing between benign or malig-nant masses. Whereas adequate preoperative imagingexaminations can clearly elucidate the extent of the re-lated tumour, which is crucial in presurgical planning.Intussusception accompanied with polyps periodicallyprotruding into the bladder is extremely rare with onlythree reported cases to our knowledge [23, 24, 28], andcould easily be misdiagnosed as bladder tumours byultrasonography.In this case, partial lobulated polyps were cauterized

using Holmium: YAG laser ureteroscopically, followedby open segmental resection of the ureter with reanasto-mosis. In the literatures, except for one case reported byF Hajji et al. which was managed by resection of thewhole polyp containing the stalk by ureteroscopic elec-trocauterization and got a following automatic resolutionof transient intussusception [28], almost all other casesof benign ureteral tumors were finally managed by surgi-cal resection of the involved ureter with reanastomosisby open or laparoscopic approach [23, 24]. We agreethat ureteroscopic cauterization is an effective and min-imally invasive treatment for the management of smallisolated ureteral polyps with mild and transient ureteralintussusception. However, for patients suffering fromstable intussusception complicated with large polyps, itis difficult to resect ureteral lesions completely by ure-teroscopic cauterization alone. Besides, because of thelimited working space and the dissatisfied laser accuracyureteroscopically, the pursuit of a perfect excision ofintussusceptum will also increase the risk of ureteralperforations, postoperative lesion recurrences, and ur-eteral strictures. Moreover, James Sewell et al. reportedthe only case of ureteral calculus-related intussusception,which was treated by ureteropyeloscopic lithotripsy withHolmium: YAG laser [26]. In that case, due to the looseand short range of the intussusceptum, ureteral intussus-ception resolved automatically following clearance of thecalculi [26]. Biopsies of the tumour and intussusceptedureter via transurethral ureteroscopy would help ruleout malignancy and signs of ischemia. The treatment

Dong et al. BMC Nephrology (2020) 21:246 Page 3 of 9

Table

1Review

ofpreviouslypu

blishe

dcasesof

ureteralintussusception

Autho

rYear

Age

/Sex

Num

ber

Aetiology

Lesion

Locatio

nLocatio

nof

pain

Hem

aturia

Hydrone

phrosis

NCC

TCEC

TCTU

IVU

Treatm

ent

Dix

[1]

1937

54/

M1

Papilloma

L/Pe-

ureter

No

Yes

Yes

N/A

N/A

N/A

Novisualizationof

left

ureter

Nep

hrou

retectom

y

Morley

etal.[2]

1952

19/

M1

Fibrou

sureteral

polyp

R/Pe-

ureter

Righ

tflank

No

Yes

N/A

N/A

N/A

Themiddleureter

isdilatedwith

filling

defects,thelower

ureter

appe

arsno

rmal

Nep

hrou

retectom

y(The

leng

thof

the

involved

ureter

istoo

long

tope

rmit

reanastomosis)

Bon

omini

etal.[3]

1963

20/

M1

Benign

pedu

nculated

polyp

L/Pe-

ureter

Leftflank

Yes

Yes

N/A

N/A

N/A

Themiddlethird

ofthe

ureter

isdilatedand

occupied

with

defects

offilling

Segm

entalresectio

nof

theureter

with

reanastomosis

Gerdes

etal.[4]

1966

34/F

1Ang

iofib

romatou

spo

lyp

N/A

Diffuse

abdo

men

Yes

No

N/A

N/A

N/A

N/A

N/A

Mazer

etal.

[5]

1979

66/

M1

TCC

R/Pe-

ureter

No

Yes

Yes

N/A

N/A

N/A

A“bell-shape

d”filling

defect

Localexcisionof

the

tumou

rwas

perfo

rmed

with

reconstructio

nof

the

ureter

Fiorelli

etal.[6]

1981

25/F

1Fibroe

pithelial

polyp

L/UPJ

Leftflank

No

Yes

N/A

N/A

N/A

Ureterald

efectand

easy

flow

ofcontrast

med

ium

arou

nditare

barelyvisible.

Resectionof

the

pyelou

reteral

junctio

nthat

was

shoved

into

distal

ureter

afterlysisof

outerfib

rous

band

was

perfo

rmed

followed

byan

And

erson-Hynes

pyelop

lasty

Vog

elzang

etal.[7]

1981

27/

M1

Fibrou

sureteral

polyp

L/Pe-

ureter

Leftflank

Yes

Yes

N/A

N/A

N/A

Mod

eratedilatatio

nin

theproxim

alureter,no

contrastpassed

into

intussusception

Tumor

excision

byureterotom

y,no

detail

Fuku

shi

etal.[8]

1983

59/F

1Inflanm

matory

Polyp

L/Pe-

ureter

Leftflank

Yes

Yes

N/A

N/A

N/A

“Claw

ofcrab

-shape

d”contrastfillingsign

Intussusceptionwas

repairedafterlysisof

theexternal

adhe

sion

ofthe

invaginatedregion

Take

uchi

etal.[9]#

1984

70/

M3

TCC(×3)

R/Ab-

ureter

No

Yes

Yes

N/A

“Con

centric

sign

”in

ureter

and

alarge

massin

bladde

r(×1)

N/A

Adilatatio

nin

the

lower

ureter

with

filling

defect

ofvariedsize,

protruding

into

bladde

r

Segm

ental

ureterectomy(×1),

combine

dwith

partialb

ladd

erresection(×

1)or

radicalcystectom

y(×

1)

71/

ML/Ab-

ureter

and

bladde

r

52/

R/Ab-

Dong et al. BMC Nephrology (2020) 21:246 Page 4 of 9

Table

1Review

ofpreviouslypu

blishe

dcasesof

ureteralintussusception(Con

tinued)

Autho

rYear

Age

/Sex

Num

ber

Aetiology

Lesion

Locatio

nLocatio

nof

pain

Hem

aturia

Hydrone

phrosis

NCC

TCEC

TCTU

IVU

Treatm

ent

Mureter

Hau

pert

etal.[10]#

1985

N/A

1Fibroe

pithelial

polyp

N/A

N/A

N/A

N/A

N/A

N/A

N/A

N/A

Segm

entalresectio

nof

theureter

with

reanastomosis

Com

pton

etal.[11]

1986

50/

M1

TCC

R/Pe-

ureter

No

Yes

No

N/A

N/A

N/A

Afusiform

dilatatio

nin

themid-ureter

Segm

entalresectio

nof

theureter

with

reanastomosis

Gab

riel

etal.[12]

1986

74/

M1

TCC

R/Pe-

ureter

Righ

tlower

abdo

men

No

Yes

N/A

N/A

N/A

Alocalw

iden

ingwith

irreg

ular

fillingde

fect

proxim

alto

the

ureterovesicaljunctio

n

Nep

hrou

reterectom

yinclud

ingacuffof

urinarybladde

r

Moretti

etal.[13]

1987

59/

M1

TCC

R/Pe-

ureter

No

Yes

Yes

N/A

N/A

N/A

Atubu

larno

nopaqu

efillingde

fect

with

indilatedlower

ureter

that

contains

apparentlycalcified

polyp

Intussusceptionwas

redu

cedafterlocal

excision

ofthe

tumou

rin

ureter

Duc

hek

etal.[14]

1987

24/

M1

Papilloma

R/Pe-

ureter

Righ

tflank

Yes

Yes

N/A

N/A

N/A

Alocalw

iden

ingin

the

middleureter

with

irreg

ular

fillingde

fect

Partialresectio

nof

theureter

with

reanastomosis

Radhi

[15]

1992

N/A

1TC

CN/A

N/A

N/A

N/A

N/A

N/A

N/A

N/A

N/A

Pnget

al.

[16]

1995

26/F

1Fibroe

pithelial

polyp

R/Ab-

ureter

Righ

tflank

Yes

Yes

N/A

N/A

N/A

Afillingde

fect

inthe

ureter

Segm

entalresectio

nof

theureter

with

reanastomosis

Bernh

ard

etal.[17]

1996

45/

M1

Latrog

enic1

L/Pe-

ureter

Leftflank

N/A

Yes

N/A

N/A

N/A

Afillingde

fect

inthe

mid-ureter

Partialresectio

nof

theureter

and

constructio

nof

aBo

ariflap

elKha

der

etal.[18]#

1997

30/F

1Latrog

enic2

-/UPJ

N/A

N/A

N/A

N/A

N/A

N/A

N/A

Segm

entalresectio

nof

theureter

with

calicou

reterostom

y

deLa

Tailleet

al.

[19]

1998

59/

M1

TCC

R/Ab-

ureter

No

Yes

Yes

Aureteral

mass

N/A

N/A

Ureteralfusiform

enlargem

ent

containing

anoval

fillingde

fect

Nep

hrou

retectom

y

Liuet

al.

[20]

2000

63/

M1

Latrog

enic3

R/Ab-

ureter

N/A

Yes

Yes

N/A

N/A

N/A

N/A

Theintussusception

was

recogn

ized

atthetim

eandwas

redu

cedcompletely

with

hydrostatic

pressure

and

perfo

rmed

with

use

offluoroscopic

Dong et al. BMC Nephrology (2020) 21:246 Page 5 of 9

Table

1Review

ofpreviouslypu

blishe

dcasesof

ureteralintussusception(Con

tinued)

Autho

rYear

Age

/Sex

Num

ber

Aetiology

Lesion

Locatio

nLocatio

nof

pain

Hem

aturia

Hydrone

phrosis

NCC

TCEC

TCTU

IVU

Treatm

ent

guidance

and

consciou

ssedatio

n.

Chion

get

al.[21]

2004

15/

M1

Latrog

enic4

R/UPJ

N/A

N/A

Yes

N/A

N/A

N/A

A“bell-shape

d”filling

defect

atthedistalpart

oftheup

perureter

Segm

entalresectio

nof

theob

structing

ureter

with

Culp-De-

Weerd

pyelop

lasty

reconstructio

n

Xuet

al.

[22]

2007

49/

M1

Fibroe

pithelial

polyp

R/Pe-

ureter

Righ

tflank

Yes

No

An

enlargem

ent

inthe

inferio

rpart

ofthe

invaginated

ureter

“Con

centric

sign

”N/A

Thewallo

fthe

intussuscepted

ureter

appe

ared

asa

cylindricalfillingde

fect

inthedilatatedureteral

lumen

.Invaginated

ureterallumen

isfilled

with

contrastmaterial

andappe

ared

asa

“line

”sign

inthe

intussusception

Segm

entalresectio

nof

theureter

with

reanastomosis

Jinet

al.

[23]

2011

63/F

1Massive

fibroep

ithelial

polyp

R/Pe-

ureter

No

Yes

No

N/A

“Con

centric

sign

”Afilling

defect

inlower

enlarged

ureter

A“spo

nge-like”

filling

defect

inlower

enlarged

ureteral

segm

entandan

“oval-

shaped

”fillingde

fect

oftheureteralorifice

inside

thebladde

r

Segm

entalresectio

nof

theureter

with

reanastomosisviaa

laparoscop

icapproach

Haseg

awa

etal.[24]

2011

39/F

1Fibroe

pithelial

Polyp

R/Pe-

ureter

Righ

tflank

No

No

Aureteral

mass

N/A

N/A

A“ton

gue-like”

filling

defect

intheureter

Segm

entalresectio

nof

theureter

with

reanastomosisviaa

retrop

erito

neoscopic

approach

Cha

oet

al.

[25]

2012

64/

M1

TCC

R/Pe-

ureter

Epigastric

No

Yes

Aureteral

mass

“Bull’s-eye

sign

”A“stalk-of-

corn”

appe

arance

oncoronal

andsagittal

imaging

N/A

Nep

hrou

retectom

y

Sewell

etal.[26]

2015

70/

M1

Calculus

L/Pe-

ureter

No

No

Yes

A8mm

calculus

N/A

N/A

A“gob

letsign

”contrast

fillingon

retrog

rade

pyelog

raph

y

Ureteropyeloscopic

litho

tripsy

with

laser

Suzu

kiet

al.[27]

2015

67/

M1

Fibroe

pithelial

polyp

R/Pe-

ureter

No

Yes

Yes

N/A

“Con

centric

sign

”Alinear

contrastin

the

invaginated

proxim

alureter

Invaginatedureteral

lumen

isfilledwith

contrastmaterialand

appe

ared

asa“line

-shaped

”sign

inthe

intussusception

Segm

entalresectio

nof

theureter

with

reanastomosis

Dong et al. BMC Nephrology (2020) 21:246 Page 6 of 9

Table

1Review

ofpreviouslypu

blishe

dcasesof

ureteralintussusception(Con

tinued)

Autho

rYear

Age

/Sex

Num

ber

Aetiology

Lesion

Locatio

nLocatio

nof

pain

Hem

aturia

Hydrone

phrosis

NCC

TCEC

TCTU

IVU

Treatm

ent

Hajjiet

al.

[28]

2019

42/

M1

Fibroe

pithelial

polyp

L/Ab-

ureter

Righ

tabdo

men

No

No

N/A

Aureteral

mass

protruding

into

bladde

r

N/A

A“bell-shape

d”filling

defect

anda“V-

shaped

”ureter

filled

with

contrastin

the

upstream

from

the

mass

Resectionof

the

polypwith

itsstalk

byureteroscopic

electrocauterisation

Thiscase

2020

56/

M1

Fibroe

pithelial

polyp

R/Ab-

ureter

Righ

tflank

and

lower

abdo

men

Yes

No

Aureteral

mass

N/A

N/A

A“spo

nge-like”

filling

defect

inlower

enlarged

ureteral

lumen

andureteral

orifice

inside

the

bladde

r

Ureteroscop

iccauterizationof

partialp

olyp

followed

byop

ensurgeryfor

segm

entalresectio

nof

theureter

with

reanastomosis

Note:

#:on

lyEn

glishab

stract

available,

NCC

Tno

n-contrast

CT,CE

CTcontrast-enh

ancedCT(onaxialimag

ing),C

TUCTurog

raph

y;Ab-ureter:abd

ominal

partof

ureter,P

e-ureter

pelvicpa

rtof

ureter,U

PJureterop

elvic

junctio

n,Lleft,R

right,TCC

Tran

sitio

nalcellcarcino

ma,N/A

notap

plicab

leLa

trog

enic

1:second

aryto

ureteroscopy

;Latrogen

ic2:

second

aryto

doub

leCHen

doprosthesis;L

atrogen

ic3;

second

aryto

percutan

eous

neph

rostom

ycatheter

exchan

ge(retrograd

eintussusception);L

atrogen

ic4:

second

aryto

percutan

eous

endo

pyelotom

yforUPJ

obstruction

Dong et al. BMC Nephrology (2020) 21:246 Page 7 of 9

approach should be altered in accordance with the asso-ciated primary lesion, the size and location of the polyp,and the occurrence of hydronephrosis [28]. Once histo-logical examination confirms the presence of a ureteralmalignant tumour, hemiuridectomy for the urinary tractis required. Ureteral intussusception owing to benignpolyps should be treated by local excision of the polypand reconstruction of the ureter to improve ureteralpatency.In conclusion, by reviewing prior cases and presenting

a typical ureteroscopic observation, we hope to increaseclinical awareness to this unique condition. Uretero-scopy is vital for diagnosis as it can offer a comprehen-sive observation to ensure the location and size of thelesions. Extensive biopsies through ureteroscopy are ne-cessary and should be recommended, by which exclud-ing the presence of ureteral malignancies seems to beeasier and less invasive. Based on that, treatment optionas ureteroscopic cauterization, or in combination withopen or laparoscopic surgical resection of segmental ur-eter can be formulated to benefit such patients.

Supplementary informationSupplementary information accompanies this paper at https://doi.org/10.1186/s12882-020-01904-8.

Additional file 1: Supplementary video. A video recorded duringureteroscopy showing the characteristics of the intussuscepted segmentof the ureter in the dilated ureteral lumen.

AbbreviationsCT: Computed tomography; IVU: intravenous urogram

AcknowledgmentsWe are grateful to this patient for participating in this study. We also like tothank nurse Rui-min Yuan for her cooperation during the operation.

DeclarationsThe authors of this manuscript have no conflicts of interest to disclose asdescribed by the BMC Nephrology.

Authors’ contributionsWe all cared for the patient and contributed to the writing of the report. YD,WZ and TF analysed and interpreted the patient data, and drafted theoriginal manuscript. LH and JZ created the images. CH devised the originalidea for the report. YD, WM and CH revised the manuscript. Written consentfor publication was obtained from the patient. All authors read andapproved the final manuscript.

FundingThe medical innovation team project of Jiangsu Province (Grant No. CXTDA-2017-48), the key research and development project of Jiangsu Province(Grant Nos. BE2019637 and BE2017635), and the outstanding medical talentproject of Xuzhou (Grant No. 22 [2017]) provided the supporting funds formanuscript writing, English editing services, and submission review.

Availability of data and materialsAll data generated or analysed during this study are included in thispublished article.

Ethics approval and consent to participateNot applicable.

Consent for publicationWritten informed consent was obtained from the patient for publication.

Competing interestsThe authors declare that they have no competing interests.

Fig. 3 Artist’s schematic line drawing representative of the underlying pathological process

Dong et al. BMC Nephrology (2020) 21:246 Page 8 of 9

Received: 27 January 2020 Accepted: 22 June 2020

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