Why the migration of peritoneal shunt catheter into the scrotum occurs more in children: A systematic literature review

Abstract

BACKGROUND

Ventriculoperitoneal shunt (VPS) catheter insertion is one of the most widely accepted surgical procedures for hydrocephalus. Migration of the end of the distal VPS catheter into the scrotum is one of the rare complications of VPS catheter insertion.

AIM

To review the demographics, clinical characteristics, and outcomes of the surgical procedures provided for the cases of migration of the distal VPS catheter into the scrotum.

METHODS

This is a systematic review of the published cases, and the literature search was performed from 1974 to June 30, 2024, to retrieve the relevant manuscripts. The cases were grouped into two. Group A included cases of migration of the distal VPS catheter into the scrotum, detected in children. Group B included the cases of migration of the distal VPS catheter into the scrotum, which was detected in adults and older people.

RESULTS

One hundred-twenty cases of migration of the distal end of VPS catheter into the scrotum were included in this study, a systematic literature review. Group A included n = 112 cases, and group B included n = 8 cases only. Three-fourths of the cases involved the right scrotum. Bilateral involvement was rare. The indication for initial VPS insertion was congenital hydrocephalus in four-fifths of the group A cases. The majority were infants at the time of initial shunt insertion. Four-fifths of the group A cases were 24 months-old or younger at the time of clinical diagnosis and treatment was provided for migration of the distal VPS catheter into the scrotum. In children, the interval from VPS insertion or shunt revision if any, to the diagnosis of the complication mentioned above was 12 months or less in four-fifths of the cases. The repositioning of migrated distal VPS catheter into the peritoneal cavity and herniotomy was preferred for the surgical procedure, and it was performed for two-thirds of group A cases.

CONCLUSION

Migration of the distal part of the VPS catheter into the scrotum is a rare complication of cerebrospinal fluid diversion via VPS catheter insertion. It was most frequent in children, and the right side of the scrotum was most often involved.

Key Words: Infants; Children; Complication; Hydrocephalus; Migration; Scrotum

Core Tip: The present manuscript is a systematic review and was carried out to review the demographics, clinical features, and outcome of the surgical therapy executed for the migration of the distal ventriculoperitoneal shunt (VPS) catheter into the scrotum. This study included n = 120 cases (n = 112 children and n = 8 adults and older individuals) treated for the complication, mentioned above, and published from 1974 to June 30, 2024. Migration of the distal VPS catheter to the right scrotum was most frequent/common, both in children and adults, and older people. In children, the majority of cases (four-fifths) were diagnosed and treated for above mentioned VPS complication during the first 24 months of their life or earlier. Additionally, the complication was detected within the first 12 months after the initial VPS insertion, in four-fifths of the cases. Clinically, the cases presented with the chief complaint of scrotal swelling of varying duration. Two-thirds of the children underwent surgical repositioning of the migrated distal VPS catheter into the peritoneal cavity and herniotomy. The systematic literature review revealed two deaths among the adults/older patients and none among the children treated for migration of the distal VPS catheter into the scrotum.

INTRODUCTION

The cerebrospinal fluid (CSF) diversion from the ventricular system to the peritoneal cavity through a ventriculoperitoneal shunt (VPS)/peritoneal shunt catheter insertion is one of the most frequently performed surgical procedures for the treatment of hydrocephalus[1-3]. However, VPS catheter insertion is technically a simple procedure associated with several complications and requires revision procedures[2-5]. The shunt complications are most frequently observed during the first 12 months after the initial shunt insertion, and it is also evidenced more in children than adults[5-7]. Migration of the distal shunt catheter into the hollow viscus (gastrointestinal tract, urinary bladder, and uterus) with or without extrusion through the natural orifices is one of the known complications of CSF shunt insertion[8-11]. Migration of the distal shunt catheter to the heart[12], pulmonary vasculature[13], retroperitoneal area, through the umbilicus, and other body parts, has also been reported in the literature[14-17]. Migration of the distal end of the peritoneal catheter into the scrotum is a rare complication of CSF shunt catheter insertion[16,17]. For reporting this manuscript, the Preferred Reporting Items for Systematic Reviews and Meta Analyses (PRISMA) guidelines are followed[18]. This report is a literature review of n = 120 cases published from 1974 to June 2024 related to the treatment of migration of the distal end of a peritoneal shunt catheter into the scrotum[19-113].

MATERIALS AND METHODS

An electronic database of PubMed, PubMed Central (PMC), Scopus, Web of Science, Cochrane Library, ResearchGate, and Google Scholar were searched to obtain the published/available literature for the distal end of the VPS/CSF shunt catheter migration into the scrotum. Ramani et al[19] in 1974 first reported a case of migration of a distal end of a peritoneal shunt catheter into the scrotum, in a 6-month-old child. The literature search was made from 1974 to June 30, 2024, to retrieve the relevant manuscripts. The keywords applied for the online search were “migration of distal VPS catheter into the scrotum”, “scrotal migration of distal VPS catheter”, “migration of distal peritoneal shunt catheter”, “migration of distal VPS catheter”, “migration of the CSF shunt catheter” and “rare/unusual migration of distal shunt catheter”. The cases/case reports with incomplete desired details were excluded from the review. Conference proceedings with cases/case reports for migration of a distal end of a VPS into the scrotum were also excluded. Cases diagnosed/treated for a migration of the distal end of a lumbar-peritoneal and a theco-peritoneal shunt catheter into the scrotum were excluded. Cases/case reports presented with extrusion of a distal end of the VPS catheter through the inguinal skin, but without migration to the scrotum were also excluded. Cases of migration of the distal CSF shunt catheter within the inguinal canal, but without migration to the scrotum were also excluded.

The guidelines of PRISMA are followed for reporting this systematic review. This is a single-author manuscript, therefore the selection of the literature, the extraction of the desired detail, paper writing, and others were carried out by the author alone. The titles, abstracts, and full texts of the manuscripts were accessed before the final selection of the manuscripts for the present review.

The details extracted from the included manuscripts were the patient’s age at the time of primary VPS insertion, the causes of hydrocephalus (indication for the primary VPS insertion), the patient’s age at the time of diagnosis of the VPS complication mentioned above, and the interval from VPS insertion/last VPS revision if any, to the diagnosis of the migration of the distal end of a peritoneal shunt catheter into the scrotum. The other details extracted from the manuscripts were the clinical characteristics, radiological diagnostic modalities used, and the surgical procedures’ outcomes.

The migration of the distal VPS catheter into the scrotum is grouped into two groups, group A and group B. Group A included the cases of migration of the distal VPS catheter into the scrotum, detected/diagnosed and treated in children. Group B included the cases of migration of the distal VPS catheter into the scrotum, detected/diagnosed and treated in adults and older people.

The surgical therapy executed for the cases of migration of the distal VPS catheter into the scrotum are grouped as below: (1) A: Reposition of migrated distal VPS catheter into the peritoneal cavity and inguinal herniotomy; (2) B: Reposition (spontaneous) of migrated distal VPS catheter into the peritoneal cavity and inguinal herniotomy; (3) C: Removal of part of migrated distal VPS catheter, repositioning of the remaining peritoneal catheter into the peritoneal cavity and inguinal herniotomy; (4) D: Removal of part of migrated distal VPS catheter, repositioning of the remaining peritoneal catheter into the peritoneal cavity without inguinal herniotomy; (5) E: Removal of migrated/abandoned distal VPS catheter with repositioning of working distal VPS catheter into the peritoneal cavity and inguinal herniotomy; (6) F: Removal of migrated/abandoned distal VPS catheter and inguinal herniotomy; (7) G: Removal of the entire VPS catheter alone; (8) H: Removal of the entire VPS catheter and inguinal herniotomy; (9) I: Removal of entire VPS catheter with re-VPS insertion/VPS revision and inguinal herniotomy; (10) J: Surgical therapy not required for the migrated distal VPS catheter; (11) K: Surgical therapy not done for the migrated distal VPS catheter; (12) L: Surgical therapy details not available for the migrated distal VPS catheter; and (13) M: Other surgical procedures for the migrated distal VPS catheter.

An ethical committee approval was not required for this manuscript, as is a systematic literature review of the already published cases/case reports.

RESULTS

One hundred twenty cases of the migration of the distal end of a VPS catheter into the scrotum were included in the study, a systematic review, and were retrieved from the n = 95 manuscripts. PRISMA flow diagram for the database search, screening, and selection of the literature/manuscripts for the present systematic review are presented in Figure 1. Group A (infants and children below the age of 18 years) included a total of n = 112 cases, detailed in Table 1[19-105]. Group B (adults and older people) included n = 8 cases, detailed in Table 2[106-113]. The year-wise details for the manuscripts included in the present review are provided in Table 3. More than half (n = 67) of the cases were published as isolated cases and are detailed in Figure 2. The year-wise details for the cases included in the present review are provided in Table 4. A total of 9 articles, including n = 12 cases, of migration of the distal VPS catheter into the scrotum, were excluded from the review, and detailed in Table 5[114-122].

Figure 1  Preferred Reporting Items for Systematic Reviews and Meta Analyses flow diagram for literature search for the systematic review of migration of the distal ventriculoperitoneal shunt catheter into the scrotum.

Figure 2  Types of literature (manuscripts) included in the systematic review (n = 95).

Table 1 Patient demographics and details of ventriculoperitoneal shunt catheter migration into the scrotum in children (n = 112).

Number	Ref.	Indication for VPS insertion	Age VPS insertion	Age scrotal migration	Interval (months)	VPS (revision)	Site1 (right/left)	Operative procedures executed	Outcome
1	Ramani et al[19], 1974	Hydrocephalus (congenital)	Neonate (12 days)	6 months	5 months	No	Right	A	R
2	Levey et al[20], 1977	Hydrocephalus with NTD (congenital)	Neonate (13 days)	1 month	6 days	Yes	Right	A	R
3	Redman et al[21], 1978	Hydrocephalus (congenital?)	N/A	6 months	N/A	No	Right	A	R
4	Bristow et al[22], 1978	Hydrocephalus (congenital)	10 months	10 months	1 day	No	Right	C	R
5	Crofford et al[23], 1983	Hydrocephalus (post haemorrhagic)	Neonate (15 days)	6 months	5 months	No	Right	A	R
6	Crofford et al[23], 1983	Hydrocephalus (congenital)	Neonate (1 month)	3 months	2 months	No	Right	A	R
7	Crofford et al[23], 1983	Hydrocephalus (congenital)	3 months	4 months	1 month	No	Right	A	R
8	Crofford et al[23], 1983	Hydrocephalus (brain tumor)	48 months	50 months	2 months	No	Left	B	R
9	Fuwa et al[24], 1984	Hydrocephalus (congenital)	Neonate (22 days)	12 months	11 months	Yes	Left	I	R
10	Ram et al[25], 1987	Hydrocephalus (post infective)	6 months	36 months	30 months	No	Right	C	R
11	Kwok et al[26], 1989	Hydrocephalus (congenital)	Neonate (1 month)	6 months	1 week	Yes	Bilateral	E	R
12	Calvário et al[27], 1990	Hydrocephalus (congenital)	Neonate (1 month)	24 months	23 months	No	Right	A	R
13	Göçer et al[28], 1990	Hydrocephalus (congenital)	2 months	10 months	8 months	No	Right	C	R
14	Selcuklu[29], 1991	Hydrocephalus with NTD (congenital)	Neonate	8 months	7 months	No	Right	B	R
15	Ammar et al[30], 1991	Hydrocephalus (congenital)	5 months	7 months	2 months	No	Left	G	R
16	Jamjoom et al[31], 1992	Hydrocephalus (congenital)	Neonate (3 weeks)	14 months	2 months	Yes	Right	E	R
17	Wong[32], 1994	Hydrocephalus (congenital)	6 months	7 months	1 month	No	Right	A	R
18	Goh et al[33], 1997	Hydrocephalus (congenital)	Neonate (1 day)	4 months	4 months	No	Right	B	R
19	Oktem et al[34], 1998	Hydrocephalus (congenital)	10 months	16 months	6 months	No	Right	A	R
20	Oktem et al[34], 1998	Hydrocephalus (congenital)	2.5 months	8 months	5 months	No	Right	A	R
21	Oktem et al[34], 1998	Hydrocephalus (congenital)	Neonate (9 days)	4 months	4 months	No	Right	A	R
22	Oktem et al[34], 1998	Hydrocephalus (congenital)	2.5 months	2.5 months	1 day	No	Left	A	R
23	Porras Estrada et al[35], 1999	Hydrocephalus (congenital)	Neonate (1 day)	11 months	5 months	Yes	Left	A	R
24	Ozveren et al[36], 1999	Hydrocephalus with NTD (congenital)	Neonate (3 days)	4 days	1 day	No	Right	A	R
25	Silver et al[37], 2000	Hydrocephalus (congenital)	6 months	36 months	N/A	Yes	Right	F	R
26	Ward et al[38], 2001	Hydrocephalus (post infective)	11 months	18 months	7 months	No	Right	A	R
27	Gupta[39], 2003	Hydrocephalus (congenital)	Neonate	10 months	9 months	No	Right	A	R
28	Henriques et al[40], 2003	Hydrocephalus (post-haemorrhagic)	Neonate	5 months	4 months	No	Right	A	R
29	Walsh et al[41], 2004	Hydrocephalus (congenital)	5 months	11 months	6 months	No	Right	L	N/A
30	de Aquino et al[42], 2006	Hydrocephalus with NTD (congenital)	Neonate (1 day)	14 months	14 months	No	Right	L	R
31	de Aquino et al[42], 2006	Hydrocephalus (congenital)	Neonate (11 days)	1 month	20 days	No	Right	L	R
32	Moon et al[43], 2007	Hydrocephalus (congenital)	4 months	12 years	4 months	Yes	Left	C	R
33	Karaosmanoglu et al[44], 2008	Hydrocephalus with NTD (congenital)	N/A	14 months	N/A	No	Right	G?	R
34	Kayaoğlu et al[45], 2008	Hydrocephalus with NTD (congenital)	Neonate (10 days)	5 months	4 months	No	Right	C	R
35	Agarwal et al[46], 2009	Hydrocephalus (congenital?)	8 months	24 months	7 months	Yes	Right	A	R
36	Rahman et al[47], 2009	Hydrocephalus (brain tumor)	48 months	49 months	1 month	No	Right	C	R
37	de Quintana-Schmidt et al[48], 2010	Hydrocephalus (congenital)	Neonate	2 months	1.6 month	No	Left	A	R
38	Kita et al[49], 2010	Hydrocephalus (brain tumor)	60 months	64 months	4 months	No	Left	A	R
39	Ho et al[50], 2011	Hydrocephalus (congenital)	N/A	14 years	12 months	Yes	Left	F	R
40	Potineni et al[51], 2012	Hydrocephalus (congenital)	2 months	2 months	1 day	No	Left	A	R
41	Mohammadi et al[52], 2012	Hydrocephalus (congenital)	7 months	12 months	5 months	No	Right	A	R
42	Alladi et al[53], 2012	Hydrocephalus with NTD (congenital)	Neonate	20 months	19 months	No	Left	J	R
43	Gupta et al[54], 2012	Hydrocephalus (congenital)	6 months	24 months	18 months	No	Right	A	R
44	Kundal et al[55], 2012	Hydrocephalus (congenital)	6 months	24 months	18 months	No	Right	A	R
45	Rivero-Garvía et al[56], 2013	Hydrocephalus (post infective)	12 months	72 months	60 months	No	Right	A	R
46	Elizabeth et al[57], 2013	Hydrocephalus (post-haemorrhagic)	2 months	14 months	12 months	No	Right	A	R
47	Ghasemi and Ashouri[58], 2013	Hydrocephalus (congenital)	2 months	9 months	7 months	No	Right	A	R
48	Shahizon et al[59], 2013	Hydrocephalus (congenital)	N/A	14 years	7 months	Yes	Left	F	R
49	Panda et al[60], 2013	Hydrocephalus (congenital)	18 months	60 months	42 months	No	Left	A	R
50	Ojo et al[61], 2013	Hydrocephalus (congenital)	10 months	15 months	5 months	No	Left	A	R
51	Erikci et al[62], 2014	Hydrocephalus (congenital)	2 months	48 months	46 months	No	Left	A	R
52	Erikci et al[62], 2014	Hydrocephalus with NTD (congenital)	Neonate (6 days)	2 months	2 months	No	Right	A	R
53	Shankar et al[63], 2014	Hydrocephalus (congenital)	Neonate (1 month)	12 months	11 months	No	Right	A	R
54	Basaran et al[64], 2014	Hydrocephalus (congenital)	Neonate (12 days)	6 months	5 months	No	Right	C	R
55	Kumar and Jagannathan[65], 2015	Hydrocephalus (congenital)	3 months	11 months	8 months	No	Left	A	R
56	Bayindir et al[66], 2015	Hydrocephalus with NTD (congenital)	3 months	18 months	2 months	Yes	Right	A	R
57	Sarangi et al[67], 2016	Hydrocephalus (post infective)	12 months	48 months	36 months	No	Left	G	R
58	Singh et al[68], 2016	Hydrocephalus (congenital)	Neonate	8 months	7 months	No	Right	A	R
59	Singh et al[68], 2016	Hydrocephalus with NTD (congenital)	7 months	14 months	7 months	No	Right	A	R
60	Ricci et al[69], 2016	Hydrocephalus (congenital?)	24 months	10 years	N/A	Yes	Left	I	R
61	Hung et al[70], 2017	Hydrocephalus (post-haemorrhagic)	3 months	5 months	2 months	No	Right	A	R
62	Javadi et al[71], 2017	Hydrocephalus (congenital)	8 months	10 months	2 months	No	Right	A	R
63	Kumar et al[72], 2017	Hydrocephalus with NTD (congenital)	Neonate (14 days)	48 months	47 months	No	Right	A	R
64	Fernandez-Ibieta et al[73], 2017	Hydrocephalus (congenital)	Neonate	7.5 months (mean)	6.5 months (mean)	No	Right	A	R
65	Fernandez-Ibieta et al[73], 2017	Hydrocephalus (congenital)	Neonate	7.5 months (mean)	6.5 months (mean)	No	Right	A	R
66	Fernandez-Ibieta et al[73], 2017	Hydrocephalus (congenital)	Neonate	7.5 months (mean)	6.5 months (mean)	No	Right	A	R
67	Fernandez-Ibieta et al[73], 2017	Hydrocephalus (congenital)	Neonate	7.5 months (mean)	6.5 months (mean)	No	Right	A	R
68	Fernandez-Ibieta et al[73], 2017	Hydrocephalus (congenital)	Neonate	7.5 months (mean)	6.5 months (mean)	No	Right	A	R
69	Fernandez-Ibieta et al[73], 2017	Hydrocephalus (congenital)	Neonate	7.5 months (mean)	6.5 months (mean)	No	Left?	A	R
70	Fernandez-Ibieta et al[73], 2017	Hydrocephalus (congenital)	Neonate	7.5 months (mean)	6.5 months (mean)	No	Left	D	R
71	Bawa et al[74], 2017	Hydrocephalus with NTD (congenital)	3 months	7 months	4 months	No	Right	A	R
72	Bawa et al[74], 2017	Hydrocephalus (congenital)	48 months	51 months	3 months	No	Right	A	R
73	Bawa et al[74], 2017	Hydrocephalus (congenital)	10 months	15 months	5 months	No	Right	A	R
74	Bawa et al[74], 2017	Hydrocephalus (congenital)	48 months	52 months	4 months	No	Right	I	R
75	Paterson et al[75], 2018	Hydrocephalus (congenital?)	11 months	13 months	1 week	Yes	Right	C	R
76	Deora et al[76], 2018	Hydrocephalus (congenital)	3 months	12 months	9 months	No	Right	A	R
77	Shah et al[77], 2018	Hydrocephalus (post infective)	13 months	30 months	17 months	No	Right	A	R
78	Nawaz et al[78], 2018	Hydrocephalus (post infective)	2 months	6 months	4 months	No	Right	A	R
79	Burhan et al[79], 2018	Hydrocephalus (congenital?)	N/A	24 months	N/A	No	Right	A	R
80	Ezzat et al[80], 2018	Hydrocephalus (congenital)	2 months	3 months	1 month	No	Left	A	R
81	Ezzat et al[80], 2018	Hydrocephalus (congenital)	2 months	3 months	1 month	No	Right?	A	R
82	Ezzat et al[80], 2018	Hydrocephalus (congenital)	Neonate (1 month)	2 months	1 month	No	Right?	A	R
83	Soufiany et al[81], 2018	Hydrocephalus (congenital)	Neonate (1 month)	6 months	7 days	Yes	Bilateral	E	R
84	Soufiany et al[81], 2018	Hydrocephalus (N/A)	56 months	60 months	4 months	No	Right	A	R
85	Dharmajaya[82], 2018	Hydrocephalus (congenital)	5 months	12 months	7 months	No	Right	A	R
86	Rajendran et al[83], 2019	Hydrocephalus (congenital)	3 months	36 months	33 months	No	Right	A	R
87	Shedrov et al[84], 2019	Hydrocephalus (congenital)	2 months	9 years	48 months	Yes	Right	F	R
88	Shedrov et al[84], 2019	Hydrocephalus (post hemorrhage)	3 months	24 months	21 months	No	Right	A	R
89	Abdoli et al[85], 2019	Hydrocephalus (congenital?)	23 months	24 months	10 days	No	Right	A	R
90	Abdoli et al[85], 2019	Hydrocephalus (congenital)	2 months	12 months	10 months	No	Right	A	R
91	Abdoli et al[85], 2019	Hydrocephalus (congenital)	5 months	17 months	12 months	No	Right	A	R
92	Abdoli et al[85], 2019	Hydrocephalus (congenital)	8 months	12 months	4 months	No	Left	A	R
93	Chan et al[86], 2019	Hydrocephalus (congenital)	Neonate (16 days)	18 months	17 months	No	Right	H	R
94	Prates Junior et al[87], 2020	Hydrocephalus (congenital)	Neonate (3 days)	1.6 months	1 month	No	Left	I	R
95	Gupta and Gupta[88], 2020	Hydrocephalus (congenital)	Neonate (1 month)	11 months	3 months	Yes	Right	A	R
96	Khaliq et al[89], 2021	Hydrocephalus with NTD (congenital)	Neonate	12 months	11 months	No	Right	A	R
97	Agarwal et al[90], 2021	Hydrocephalus (post infective)	15 months	18 months	3 months	No	Right	D	R
98	Javed et al[91], 2021	Hydrocephalus (congenital)	5 months	5 months	4 days	No	Right	A	R
99	Hauser et al[92], 2021	Hydrocephalus with NTD (congenital)	Neonate (3 weeks)	23 months	22 months	No	Right	B	R
100	Ahmed et al[93], 2021	Hydrocephalus (congenital)	Neonate (1 month)	8 months	7 months	No	Left	A	R
101	Alkhudari et al[94], 2022	Hydrocephalus (post-haemorrhagic)	Neonate?	6 months	5 months?	No	Right	J	R
102	Taha et al[95], 2022	Hydrocephalus with NTD (congenital)	Neonate (1 month)	3 months	2 months	No	Right	A	R
103	Topp et al[96], 2023	Hydrocephalus with NTD (congenital)	Neonate (6 days)	16 months	16 months	No	Right	A	R
104	Tzeng et al[97], 2023	Hydrocephalus (post-haemorrhagic)	Neonate?	2 months?	1 month	No	Right	K	R
105	Chettahi et al[98], 2023	Hydrocephalus (congenital)	Neonate (1 month)	60 months	59 months	No	Right	A	R
106	Joseph et al[99], 2023	Hydrocephalus (congenital)	Neonate (12 days)	6 months	1 month	Yes	Left	K	??
107	Chanchlani et al[100], 2023	Hydrocephalus (congenital)	2 months	2 months	5 days	No	Right	A	R
108	Fitra and Asrizal[101], 2023	Hydrocephalus (congenital)	Neonate (1 month)	1.5 months	2 weeks	No	Left	J	R
109	Kiyak and Cansaran[102], 2023	Hydrocephalus with NTD (congenital)	1.5 months	4 months	2 months	No	Left	B	R
110	Darwish et al[103], 2023	Hydrocephalus (congenital)	Neonate (1 month)	6 months	5 months	No	Right	A	R
111	Castrillo et al[104], 2023	Hydrocephalus (congenital)	Neonate (1 month)	3 months	2 months	No	Right	B	R
112	Javeed et al[105], 2024	Hydrocephalus with NTD (congenital)	6.5 months	7 months	13 days	No	Right	A	R

¹Site: Site of migration.

In tables the cases are listed as in the order of publication i.e.: The case published first in 1974 as first case in table. A: Reposition of migrated distal ventriculoperitoneal shunt catheter into the peritoneal cavity and inguinal herniotomy; B: Reposition (spontaneous) of migrated distal ventriculoperitoneal shunt catheter into the peritoneal cavity and inguinal herniotomy; C: Removal of part of migrated distal ventriculoperitoneal shunt catheter, repositioning of the remaining peritoneal catheter into the peritoneal cavity and inguinal herniotomy; D: Removal of part of migrated distal ventriculoperitoneal shunt catheter, repositioning of the remaining peritoneal catheter into the peritoneal cavity without inguinal herniotomy; E: Removal of migrated/abandoned distal ventriculoperitoneal shunt catheter with repositioning of working distal ventriculoperitoneal shunt catheter into the peritoneal cavity and inguinal herniotomy; F: Removal of migrated/abandoned distal ventriculoperitoneal shunt catheter and inguinal herniotomy; G: Removal of the entire ventriculoperitoneal shunt catheter alone; H: Removal of the entire ventriculoperitoneal shunt catheter and inguinal herniotomy; I: Removal of entire ventriculoperitoneal shunt catheter with re-ventriculoperitoneal shunt insertion/ventriculoperitoneal shunt revision and inguinal herniotomy; J: Surgical therapy not required for the migrated distal ventriculoperitoneal shunt catheter; K: Surgical therapy not done for the migrated distal ventriculoperitoneal shunt catheter; L: Surgical therapy details not available for the migrated distal ventriculoperitoneal shunt catheter; N/A: Not available/details not available; NTD: Neural tube defect; R: Recovered; VPS: Ventriculoperitoneal shunt.

Table 2 Patient demographics and details of ventriculoperitoneal shunt catheter migration in adults and older people (n = 8).

Number	Ref.	Indication for VPS insertion	Age VPS insertion	Age scrotal migration	Interval	VPS (revision)	Site (right/left)	Operative procedures executed	Outcome
1	Rehm et al[106], 1997	Hydrocephalus (brain tumor)	46 years	50 years	4 years	No	Right	G	Death
2	Korfias et al[107], 2013	Hydrocephalus (arachnoid cyst)	29.6 years	30 years	6 months	No	Left	A	R
3	Lee et al[108], 2015	Hydrocephalus (NPH)	65 years	65 years	7 days	No	Right	D	R
4	Foster et al[109], 2019	Hydrocephalus (post haemorrhagic)	Infant? (N/A)	27 years	Days?	Yes	Right	B	R
5	Perret et al[110], 2021	Hydrocephalus (N/A)	16 years	22 years	6 years	No	Right	I	R
6	Khoudir et al[111], 2022	Hydrocephalus (NPH)	75 years	75.4 years	4 months	No	Right	C	R
7	Ndongo Sonfack et al[112], 2022	Hydrocephalus (NPH)	73 years	73 years	2 days	Yes	Right	A	R
8	Vicaría and Unzué[113], 2022	Hydrocephalus (NPH)	80 years	80.2 years	47 days	No	Right	G	Death

A: Reposition of migrated distal ventriculoperitoneal shunt catheter into the peritoneal cavity and inguinal herniotomy; B: Reposition (spontaneous) of migrated distal ventriculoperitoneal shunt catheter into the peritoneal cavity and inguinal herniotomy; C: Removal of part of migrated distal ventriculoperitoneal shunt catheter, repositioning of the remaining peritoneal catheter into the peritoneal cavity and inguinal herniotomy; D: Removal of part of migrated distal ventriculoperitoneal shunt catheter, repositioning of the remaining peritoneal catheter into the peritoneal cavity without inguinal herniotomy; G: Removal of the entire ventriculoperitoneal shunt catheter alone; I: Removal of entire ventriculoperitoneal shunt catheter with re-ventriculoperitoneal shunt insertion/ventriculoperitoneal shunt revision and inguinal herniotomy; N/A: Not available/details not available; NPH: Normal pressure hydrocephalus; R: Recovered; VPS: Ventriculoperitoneal shunt.

Table 3 Yearly overview of literature included in the systematic review (n = 95).

Publication (year-wise)	Group A	Group B	Total (A+B)	Standard deviation	Mean	95%CI (margin of error)
1974–1980	4	0	4	2	2	2-2.772 (± 138.59%)
1981–1985	2	0	2	1	1	1-1.386 (± 138.59%)
1986–1990	4	0	4	2	2	2-2.772 (± 138.59%)
1991–1995	4	0	4	2	2	2-2.772 (± 138.59%)
1996–2000	5	1	6	2	3	3-2.772 (± 92.40%)
2001–2005	4	0	4	2	2	2-2.772 (± 138.59%)
2006–2010	8	0	8	4	4	4-5.544 (± 138.59%)
2011–2015	17	2	19	7.5	9.5	9.5-10.394 (± 109.42%)
2016–2020	22	1	23	10.5	11.5	11.5-14.552 (± 126.54%)
2021–2024	17	4	21	6.5	10.5	10.5-9.009 (± 85.80%)
Total	87	8	95	39.5	47.5	47.5-54.744 (± 115.25%)

Table 4 Yearly overview of cases included in the systematic literature review (n = 120).

Publication (year-wise)	Group A cases	Group B cases	Total (A+B)	Standard deviation	Mean	95%CI (margin of error)
1974–1980	4	0	4	2	2	2-2.772 (± 138.59%)
1981–1985	5	0	5	2.5	2.5	2.5-3.465 (± 138.59%)
1986–1990	4	0	4	2	2	2-2.772 (± 138.59%)
1991–1995	4	0	4	2	2	2-2.772 (± 138.59%)
1996–2000	8	1	9	3.5	4.5	4.5-4.851 (± 107.79%)
2001–2005	4	0	4	2	2	2-2.772 (± 138.59%)
2006–2010	9	0	9	4.5	4.5	4.5-6.237 (± 138.59%)
2011–2015	18	2	20	8	10	10-11.087 (± 110.87%)
2016–2020	39	1	40	19	20	20-26.333 (± 131.66%)
2021–2024	17	4	21	6.5	10.5	10.5-9.009 (± 85.80%)
Total	112	8	120	52	60	60-72.068 (± 120.11%)

Table 5 Literature excluded from the systematic review (n = 9).

Ref.	Number of cases	Literature retrieved	Reason for exclusion
Cooper et al[114], 1976	1	Abstract	Full text not retrieved
Villarejo et al[115], 1980	1	Abstract	Full text not retrieved
Kobayashi et al[116], 1987	2	Abstract	Full text not retrieved
Albala et al[117], 1989	1	Abstract	Full text not retrieved
Celik et al[118], 2005	1	Full text	Incomplete case details
Aly et al[119], 2015	3	Full text	Incomplete case details
Vagholkar et al[120], 2016	1	Full text	Incomplete case details
Montasr[121], 2020	1	Abstract	Incomplete case details
Wu et al[122], 2024	1	Abstract	Full text not retrieved

Figure 3 represents the site of migration of the distal VPS catheter into the scrotum in children. It was most frequently observed on the right side of the scrotum, not only in group A cases but also in group B cases. For group A cases (infants and children), the indications (causes of hydrocephalus) for the initial/primary VPS catheter insertion are detailed in Figure 4. In four-fifths of the group A cases, the indication for the primary VPS insertion was congenital hydrocephalus. For group A cases, the age of children at the time of primary VPS insertion is detailed in Figure 5. In, more than four-fifths of children (n = 96), the initial VPS catheters were implanted during infancy. For group A cases, the age of children at the time of diagnosis and the treatment provided for the scrotal migration of the distal VPS catheter are provided in Figure 6. In four-fifths of the children, their age was ≤ 24 months at the time of clinical diagnosis and treatment carried out for the VPS complication mentioned above. For group A cases, the interval from the primary VPS insertion or last VPS revision if any, to the diagnosis of the above-described CSF shunt complication is detailed in Figure 7. In more than three-fourths (n = 78) of children, this VPS complication was detected within 12 months after the initial VPS insertion/last shunt revision.

Figure 3  In children, the side of migration of the distal ventriculoperitoneal shunt catheter into the scrotum (n = 112).

Figure 4 In children, the indication (causes of hydrocephalus) for primary ventriculoperitoneal shunt insertion (n = 112). NTD: Neural tube defect.

Figure 5 In children, their age at the time of primary ventriculoperitoneal shunt catheter insertion (n = 112). VPS: Ventriculoperitoneal shunt.

Figure 6 In children, their age at the time of diagnosis of migration of the distal ventriculoperitoneal shunt catheter into the scrotum (n = 112). VPS: Ventriculoperitoneal shunt.

Figure 7 In children, the interval from primary ventriculoperitoneal shunt catheter insertion (revision, if any) to the diagnosis of migration of the distal ventriculoperitoneal shunt catheter into the scrotum (n = 112). VPS: Ventriculoperitoneal shunt.

For group B (adults and older people) cases, the mean age at the time of diagnosis of the VPS complication mentioned was 52.7 years and ranged from 22 years to 80 years. For group B cases, the indication for initial VPS catheter placement was normal pressure hydrocephalus (NPH) for fifty percent of the cases. For group B cases, the interval was 6 months or less for three- fourths (n = 6) cases and ranged from 2 days to 6 months.

The chief complaint for the entire group A and group B cases was the scrotal swelling of variable duration, which occurred within a day to a few months. In 40% (n = 45) of children, the duration of the symptoms ranged from a day to 30 days. Clinical examination of the entire group A and group B cases revealed that a tube-like structure (distal VPS catheter) was detected in the scrotum. None of the group A and group B cases had the classical features of meningitis. Clinical features of peritonitis were also absent in children, while one of group B (80-year-old) cases had features of peritonitis and scrotal abscess.

A plain Roentgenogram of the abdomen including the inguinoscrotal area, confirmed the presence of a distal VPS catheter into the scrotum in all the cases. Various other diagnostic modalities ordered before the operative therapy for VPS complication mentioned above were cranial computed tomography (CT) scan n = 16 for group A cases and n = 4 for group B cases and CT scan of the abdomen including the scrotum n = 2 for group A cases and n = 3 for group B cases.

The various surgical procedures performed for the treatment of the above-mentioned VPS complication for group A cases are detailed in Figure 8. In two-thirds (n = 73) of children, repositioning of migrated distal VPS catheter back into the peritoneal cavity and inguinal herniotomy were carried out. For group B cases, the surgical procedures performed by the authors were not uniform. Two of the cases were managed by the removal of their entire VPS catheter, and another n = 2 of the cases was managed by repositioning of migrated distal VPS catheter back into the peritoneal cavity and inguinal herniotomy. A comparison of the results obtained for groups A and B is detailed in Table 6. This present systematic review revealed n = 2 deaths among group B and none from group A cases.

Figure 8 In children, surgical procedures executed for migration of the distal ventriculoperitoneal shunt catheter into the scrotum (n = 112). A: Reposition of migrated distal ventriculoperitoneal shunt catheter into the peritoneal cavity and inguinal herniotomy; B: Reposition (spontaneous) of migrated distal ventriculoperitoneal shunt catheter into the peritoneal cavity and inguinal herniotomy; C: Removal of part of migrated distal ventriculoperitoneal shunt catheter, repositioning of the remaining peritoneal catheter into the peritoneal cavity and inguinal herniotomy; D: Removal of part of migrated distal ventriculoperitoneal shunt catheter, repositioning of the remaining peritoneal catheter into the peritoneal cavity without inguinal herniotomy; E: Removal of migrated/abandoned distal ventriculoperitoneal shunt catheter with repositioning of working distal ventriculoperitoneal shunt catheter into the peritoneal cavity and inguinal herniotomy; F: Removal of migrated/abandoned distal ventriculoperitoneal shunt catheter and inguinal herniotomy; G: Removal of the entire ventriculoperitoneal shunt catheter alone; H: Removal of the entire ventriculoperitoneal shunt catheter and inguinal herniotomy; I: Removal of entire ventriculoperitoneal shunt catheter with re-ventriculoperitoneal shunt insertion/ventriculoperitoneal shunt revision and inguinal herniotomy; J: Surgical therapy not required for the migrated distal ventriculoperitoneal shunt catheter; K: Surgical therapy not done for the migrated distal ventriculoperitoneal shunt catheter; L: Surgical therapy details not available for the migrated distal ventriculoperitoneal shunt catheter; M: Other surgical procedures for the migrated distal ventriculoperitoneal shunt catheter; VPS: Ventriculoperitoneal shunt.

Table 6 Comparison of results for group A and group B cases, n (%).

Details	Description	Group A	Group B	Total (A+B)	Standard deviation	Mean	95%CI (margin of error)
Cases	Total	112 (93.33)	8 (6.66)	120 (100)	52	60	60-72.068 (± 120.11%)
Location	Right side	83 (69.16)	7 (5.83)	90 (75)	38	45	45-52.665 (± 117.03%)
	Left side	27 (22.5)	1 (0.83)	28 (23.33)	13	14	14-18.017 (± 128.69%)
	Bilateral	2 (1.66)	0 (00)	2 (1.66)	1	1	1-1.386 (± 138.59%)
Indication (ventriculoperitoneal shunt insertion)	Congenital pathologies	94 (78.33)	0 (00)	94 (78.33)	47	47	47-65.139 (± 138.59%)
	Post-hemorrhagic	7 (5.83)	1 (0.83)	8 (6.66)	3	4	4-4.158 (± 103.94%)
	Brain tumor	3 (2.5)	2 (1.66)	5 (4.16)	0.5	2.5	2.5-0.693 (± 27.72%)
	Normal pressure hydrocephalus	0 (00)	4 (3.33)	4 (3.33)	2	2	2-2.772 (± 138.59%)
Interval	0-12 months	88 (73.33)	6 (5)	94 (78.33)	41	47	47-56.823 (± 120.90%)
	13-24 months	10 (8.33)	0 (00)	10 (8.33)	5	5	5-6.93 (± 138.59%)
Surgical procedure executed	Group A	73 (60.83)	2 (1.66)	75 (62.5)	35.5	37.5	37.5-49.2 (± 131.20%)
	Group B	6 (5)	1 (0.83)	7 (5.83)	2.5	3.5	3.5-3.465 (± 98.99%)
	Group C	8 (6.66)	1 (0.83)	9 (7.5)	3.5	4.5	4.5-4.851 (± 107.79%)
Mortality	Postoperative	0 (00)	2 (1.66)	2 (1.66)	1	1	1-1.386 (± 138.59%)

DISCUSSION

VPS insertion is preferred over ventriculoatrial shunt insertion for the treatment of hydrocephalus, not only in infants and children but also in adults and older people[123]. VPS catheter insertion is associated with various complications ranging from infection, shunt malfunction, shunt obstruction, shunt disconnection[123-125], and shunt catheter migration to the different body parts[126], and through natural orifices, and are more frequently documented in children than adults[8-11]. Many VPS complications[14-17] may require shunt revisions[123-126].

Summary of evidence

Ninety-five manuscripts were identified and included in the present systematic review of the management of migration of distal VPS catheters into the scrotum and extracted from the literature published from 1974 to June 2024[19-113]. Among a total of n = 95 manuscripts, only n = 8 was published and are available in languages other than English[35,40,43,48,56,58,84,104]. For n = 8 of the above manuscripts the abstracts are available in English, and the other parts of the manuscripts were translated into English using the “Google Translate” for extracting the desired information. The remaining n = 87 manuscripts are published and available in English[19-34,36-39,41,42,44-47,49-55,57,59-83,85-103,105-113]. Ninety-four of the manuscripts included in this review are full-text articles. Only one of the manuscripts included in this review is the abstract of a case report[32].

Further review of the literature included for the systematic review, revealed that more than two-thirds (n = 67) of manuscripts, which included a total of n = 67 cases of the migration of distal VPS catheter into the scrotum, were published as case report and detailed the isolated cases[19-22,24,25,27-33,35,36,38-40,43,45-47,49-54,56,58,60,61,64-67,69,70,72,75,76,78,82,83,86,89-91,93-106,108,110-113]. Six manuscripts, which included n = 24 cases of VPS complication described above, were published as case series[23,34,55,73,74,85]. Manuscripts that included more than two cases of VPS complications described above were considered/counted as case series[23,34,73,74,85]. Only one of the cases was extracted from a manuscript published as a case series[55]. Three of the articles, which included details for n = 6 cases, described two cases of the VPS complication in each manuscript and were also published as case reports[62,68,84]. Two of the literature that were published and available as original articles, which included n = 4 cases of VPS complication described[79,80]. Four of the manuscripts contained details of n = 6 cases and were published as review articles[42,81,87,92]. The remaining n = 13 of the manuscripts, which included n = 13 cases, were published and available under the headings of medical images[41,57,59,63,109], letters to the editor[44,48,71,77,88,107], and correspondence or communication[26,37].

A total of 95 articles were included in the systematic review of which 92% (n = 87) of the manuscripts were related to the VPS catheter migration into the scrotum, which occurred in children. A further literature review confirmed that two-thirds (n = 63) of these manuscripts were published in the past 14 years, from 2011 to June 2024. Although the first case of migration of the distal VPS catheter into the scrotum was reported in 1974, in a child, there were only n = 23 such cases reported in children between 1974 and 2005. This VPS complication is rarer in adults and older individuals, with only n = 8 cases published as of June 2024. A deeper review of the literature found that two-thirds (n = 81, group A = 74, and group B = 7) of the migration of distal VPS catheters into the scrotum were reported/published during the past 14 years from 2011 to June 2024. Half (n = 61) of the cases (both groups) were published during the past 9 years, from 2016 to June 2024. In children, only n = 25 such cases were reported/published between 1974 and 2000. Nine manuscripts, including n = 12 cases of the VPS complication described above, were excluded from this review due to incomplete case information or inability to retrieve the full texts of the articles[114-122].

In children, migration of distal VPS catheter to the right side of the scrotum was more common and evidenced in three-fourths (n = 83) of cases. The shunt catheter was migrated into the left side of the scrotum in one-fourths (n = 27) of children[23,24,30,34,35,43,48-51,53,59-62,65,67,69,73,80,85,87,93,99,101,102]. Migration of VPS catheter to both sides of the scrotum (bilateral migration) was rare and only observed in two children[26,81]. In adults and older people, the shunt catheter was migrated into the right side of the scrotum in n = 7 cases[106,108-113]. In one case, the shunt catheter was migrated into the left side of the scrotum[107].

The indications for primary VPS catheter insertion in children were most frequently for congenital hydrocephalus while in adults and older people, it was NPH in half of the cases. For group A cases (children), the indications (causes of hydrocephalus) for the initial/primary VPS catheter insertion in order of frequency were; congenital hydrocephalus/hydrocephalus of congenital origin (n = 76; 67.8%), hydrocephalus associated with neural tube defects (n = 18; 16.07%), post-infective hydrocephalus (n = 7; 6.25%), hydrocephalus developed after the ventricular hemorrhage (n = 7; 6.25%), hydrocephalus developed due to brain tumor (n = 3), and for one child the cause was not mentioned/not available. For group B cases, the indications (causes of hydrocephalus) for the primary VPS catheter insertion were NPH in half (n = 4) of the cases, followed by the hydrocephalus secondary to the brain tumor (n = 1), arachnoid cyst (n = 1), post-hemorrhagic hydrocephalus (n = 1), and the cause for hydrocephalus was not known for one case.

The initial/primary VPS catheter insertions were carried out during infancy in more than four-fifths (n = 96) of children. Of the above, at n = 49 occasions it was implanted during the neonatal period. In n = 5 children their age was 13 months to 24 months at the time of VPS catheter insertion, and for n = 6 children their age ranged from 25 months to 60 months, at the time of VPS catheter insertion. The mean age at initial VPS catheter insertion was 55.28 years for group B cases.

In more than four-fifths (n = 91) of children (group A), their age was 0 day to 24 months at the time of diagnosis and the treatment provided for the presence of the distal VPS catheter into the scrotum. In n = 67 (60%) children, their age was 0 day to 12 months. For group B cases, the mean age at the time of diagnosis of VPS complication mentioned above was 52.7 years, ranging from 22 years to 80 years[106-113].

For group A cases (children), in more than three-fourths (n = 88) cases the interval from the primary VPS insertion/last VPS revision if any, to the diagnosis of the migration of the VPS catheter into the scrotum was ≤ 12 months. In one-fourths (n = 23) of children, the VPS complication occurred within a month of VPS insertion. The VPS complication described was not frequent after 36 months of shunt implantation/shunt revision, and it was documented only in n = 6 cases[56,60,62,72,84,98]. For group B cases, the interval ranged from 2 days to 6 years. In n = 6 of group B cases, it was ≤ 6 months[107-109,111-113]. In n = 2 of group B cases, it occurred 4 years and 6 years after the VPS catheter insertion[106,110].

For the entire group A and group B cases, the chief complaint was the scrotal swelling after the VPS catheter insertion or VPS revision, if any, and scrotal swelling occurred within a day to a few months. In 40% (n = 45) of children, the symptom’s duration ranged from 1 day to 30 days. In the entire group A and group B cases, the clinical examination revealed that the distal VPS catheter was detected/palpated in the involved side of the scrotum. The classical signs of meningitis were not detected in any of the group A and group B cases. Clinical signs of peritonitis were also absent in children, while one of group B (80-year-old) cases had features of peritonitis and scrotal abscess. For confirmation of the clinical diagnosis, a plain Roentgenogram of the abdomen including the inguinoscrotal area/X-Ray series was mostly carried out for the cases, and that confirmed the presence of a distal VPS catheter into the involved scrotum in all the cases. Additionally, before surgical therapy, cranial CT scan was ordered for n = 16 children, and n = 4 of group B cases. CT scan of the abdomen including the inguinoscrotal area was ordered for n = 2 children, and n = 3 of group B cases.

In children, for migration of distal VPS catheter into the scrotum, the preferred surgical procedure was the repositioning of the migrated distal VPS catheter back into the peritoneal cavity and inguinal herniotomy, and it was carried out for two-thirds (n = 73) cases. In n = 8 children, the part of the distal shunt catheter was removed (cut short), the remaining peritoneal catheter was repositioned back into the peritoneal cavity, and an inguinal herniotomy was carried out[22,25,28,43,45,47,64,75]. In two group B cases, the repositioning of migrated distal VPS catheter back into the peritoneal cavity, and inguinal herniotomy were carried out[107,112]. In another n = 2 group B case, the entire VPS catheter was removed without inguinal herniotomy[106,113].

VPS catheter disconnection/fracture was documented in n = 8 cases, seven among children and one in a 22-year-old person[24,26,50,53,59,69,81,110]. In two children, there was the migration of the VPS shunt catheters into both sides of the scrotum, on one side it was an abandoned/disconnected peritoneal catheter, and on another side, it was a working distal VPS catheter. For both children, the abandoned/disconnected peritoneal catheter was removed, the working peritoneal catheter repositioned back into the peritoneal cavity, and bilateral inguinal herniotomies were carried out[26,81]. In another two children, the entire VPS catheter (including the disconnected peritoneal catheter) was removed and re-VPS insertion/immediate VPS revision, and inguinal herniotomy were carried out[24,69]. In a 22-year-old person, the VPS catheter was removed, and immediate VPS revision and inguinal hernia repair were carried out for the migrated VPS catheter[110].

The present systematic review revealed two deaths among group B cases[106,113]. A 46-year-old person was diagnosed with an intraventricular tumor (ependymoma) causing hydrocephalus and was operated upon (tumor excision and VPS catheter insertion). Because of severe brain damage, he was in a persistent vegetative condition. Four years after the above surgical procedures, his distal VPS catheter migrated into the right scrotum and extruded/eroded out through the scrotal skin, and was treated by removal of the entire VPS catheter. A new VPS catheter was inserted once the CSF culture became sterile. He died of poor general condition during the treatment[106]. A VPS catheter was inserted for NPH in an 80-year-old person. His distal shunt catheter was migrated into the right scrotum and treated by removal of the entire VPS catheter. He was also diagnosed with a scrotal abscess, developed peritonitis, and died within a few hours during the management[113]. There were no deaths among the n = 112 children, treated for migration of the VPS shunt catheter into the scrotum.

Following are the possible factors responsible for the migration of the distal VPS catheter into the scrotum more in infants and young children, than the older children, adults, and older people. The shunt catheter also migrated more often into the right scrotum: (1) Persistent processus vaginalis; (2) Infants and children younger than 24 months; (3) Long length of an intra-abdominal part of the peritoneal part of the VPS catheter; (4) Increasing intra-abdominal pressure due to diversion of CSF into the peritoneal cavity; and (5) VPS revision in the past.

The present review also revealed that the persistence of processus vaginalis (either as patent process vaginalis or as an inguinal hernia) was evidenced during the operative procedures carried out for migration of the distal VPS catheter into the scrotum in two-thirds of infants, and half of the younger children (13-24 months of age). This finding further supports the theory that persistent processus vaginalis is one of the important factors responsible for the VPS complication described above. In general, processus vaginalis remains patent in 80% of the children at birth, and the patency decreases with the advancing age[127]. Twenty percent of male children may have a patent processus vaginalis at the age of 24 months. Chen et al[128] in 2011 reported their findings that children who received VPS for the treatment of hydrocephalus at the age ≤ 5 years, were more prone to the development of inguinal hernia, more so during the first two years after the VPS catheter insertion[128]. In a Cohort study, Wu et al[129] included n = 1568 children aged 0 day to 5 years who received VPS catheters and were followed for the development of an inguinal hernia. They also concluded that an inguinal hernia is more likely to develop in neonates after VPS surgery than in infants, toddlers, and preschool-aged children[129].

In 60% (n = 67) of the cases, the VPS complication occurred during infancy (0-12 months), and in another one-fourths (n = 24) of cases, their age was 13-24 months at the time of diagnosis/treatment of migration of the distal VPS catheter into the scrotum. These findings correlate to the findings of patent processus vaginalis more during infancy, than older age. The insertion of a long length of the peritoneal part of the VPS catheter into the peritoneal cavity may be one of the factors responsible for the scrotal migration of the VPS catheter, as this complication was documented more in children than adults.

Diversion of CSF into the peritoneal cavity via a VPS catheter insertion may lead to accumulation of CSF into the peritoneal cavity. Excess CSF in a small peritoneal cavity in neonates and infants may further elevate the intra-abdominal pressure. The presence of CSF in the peritoneal cavity may lead to or help in increasing intra-abdominal pressure and may prevent obliteration of processus vaginalis or result in patent processus vaginalis. In a retrospective study of n = 430 children who received VPS catheters for the treatment of hydrocephalus and were followed for the development of inguinal hernia and hydrocele. The authors concluded that the raised intra-abdominal pressure may be the reason for the high occurrence of inguinal hernia and hydrocele after the insertion of the VPS catheter in these children[130].

It is not clear whether history of prior shunt revision, is also a factor responsible for the migration of shunt catheter into the scrotum. Although, the present literature review documented that n = 17; 15.1% of children had shunt revision before the shunt migration[20,24,26,31,35,37,43,46,50,59,66,69,75,81,84,88,99]. In the present review of n = 112 children diagnosed and treated for migration of shunt catheter into the scrotum, n = 83; 74% of the cases the migration was into the right scrotum, n = 27; 24% of shunt catheters migrated into the left side of the scrotum, and only two children had migration of VPS shunt catheters into both the scrotum. The above findings are probably related to the fact that in boys, approximately 60% of inguinal hernias are right-sided, 30% of them involve the left side and only 10% are bilateral inguinal hernias[127].

Comparing the results obtained from group A cases to group B cases revealed that, migration of the distal VPS catheter into the scrotum was significantly more frequently reported in children than adults and older individuals. Results further support that the younger the age of children the higher the chances of shunt catheter migration into the scrotum, as four-fifths (n = 91) of children were ≤ 24 months old, at the time of diagnosis of the VPS complication mentioned above. Hydrocephalus of congenital pathologies was the indication for primary VPS catheter insertion in four-fifths (n = 94) of children, while the most common indication for VPS catheter insertion in adults and older people was NPH and it was documented in fifty percent of the group B cases. In three-fourths of group A and group B cases, the aforementioned VPS complication was diagnosed within 12 months of shunt catheter insertion. The most preferred surgical therapy offered in children was repositioning of the migrated VPS catheter back into the peritoneal cavity and inguinal herniotomy, and it was opted by the various authors for n = 73 (65%) children. The above said surgical technique was opted only for two of the cases for group B cases. A further literature review documented n = 2 deaths, both the cases belong to group B, and there was no mortality among group A cases. One of the patients died of poor general condition, while another one died of peritonitis, caused by VPS complications. Both the cases died of either poor general condition or peritonitis, and none of them died of surgical techniques opted /surgical procedures executed.

Limitations

The present study is a systematic review of already published literature and has limitations. Nine manuscripts (n = 3 full-text, and n = 6 abstract) including n = 12 cases of migration of the distal VPS catheter into the scrotum were excluded from the review, due to either incomplete case information or unavailability of the full text of the articles[114-122]. A limited number of literature/articles were retrieved through PubMed and PMC search. Therefore, other websites such as Scopus, Web of Science, Google Scholar, and ResearchGate were also utilized for the retrieval of manuscripts.

CONCLUSION

Migration of the distal VPS catheter into the scrotum is one of the rare complications of CSF diversion executed by the VPS catheter insertion for hydrocephalus. It was more frequently documented in infants and young children, although reported also in older children, adults, and older people. The migration of the shunt catheter into the right scrotum was more often, not only in children but also in adults and older people. In children, four-fifths of children’s age was 0-24 months, at the time of diagnosis and the treatment provided for the VPS complication described above. This VPS complication was diagnosed/occurred within ≤ 12 months after the primary VPS insertion or last VPS revision if any, in more than three-fourths of children. Two-thirds of children were treated by repositioning the migrated distal VPS catheter back into the peritoneal cavity and inguinal herniotomy.