ERN eUROGEN Guidelines on the Management of Anorectal Malformations Part III: Lifelong Follow-up and Transition of Care

Abstract

Introduction Anorectal malformations (ARMs) are complex congenital anomalies of the anorectal region, oftentimes also affecting the genitourinary system. Although successful surgical correction can often be achieved in the neonatal period, many children will experience functional problems in the long term. The European Reference Network for rare and complex urogenital conditions (eUROGEN) assembled a panel of experts to address these challenges and develop comprehensive guidelines for the management of ARM.

Methods The Dutch Quality Standard for ARM served as the foundation for the development of guidelines applicable on a European level. Literature was searched in Medline, Embase, and Cochrane. The ADAPTE method was utilized to incorporate the newest available evidence. A panel of 15 experts from 7 European countries assessed currency, acceptability, and applicability of recommendations. Recommendations from the Dutch Quality Standard were adapted, adopted, or rejected and recommendations were formed considering the current evidence and/or expert consensus.

Results Lifelong follow-up, integration, and transition of care were assessed. A total of eight new studies were identified. The panel adapted 18 recommendations, adopted 6, and developed 6 de novo. Overall, the level of evidence was considered low.

Conclusion Successful lifelong follow-up and transition of care require a dedicated team of pediatric and adult specialist and an individually tailored patient-centered approach. This guideline summarizes the best available evidence on follow-up of ARM patients and provides guidance for the development of structured transition programs.

Keywords

ARM - adulthood - follow-up - transitional care - consensus statement

Introduction

Anorectal malformations (ARMs) are complex congenital anomalies of the anorectal region oftentimes also affecting the genitourinary system.[1] Although most ARMs can be successfully corrected surgically in the neonatal period, many children will experience functional problems in the long term.[2] [3] [4] In that context, transition from pediatric to adult care becomes increasingly important. The complexity and variability of the disease and associated malformations as well as lack of standardized care translate into risk of colorectal, urological, gynecological, and sexual dysfunctions.[4] [5] This can in turn result in impairment of psychosocial well-being and impaired quality of life in adolescence and adulthood.[3] [6] [7] [8]

Currently, recommendations for lifelong follow-up and a structured program for transition of care of ARM patients applicable on a European level are lacking. To provide guidance in this matter, the European Reference Network (ERN) for rare and complex urogenital conditions (eUROGEN) convened a panel of experts (the Adoption and Adaptation Working Group, AAWG) with the aim to develop comprehensive guidelines for the management of ARM. To date, 56 healthcare providers in 20 different countries are part of ERN eUROGEN, and together they care for approximately 12,500 patients each year.[9] The Dutch “Quality Standard for Anorectal Malformations” of 2020 served as the basis for an adaptation process of the guidelines on a European level, which was performed according to the ADAPTE method.[10] [11] [12]

The adaptation report comprises six categories: diagnostics, treatment, organization of care, collaboration, referral and communication between providers, lifelong follow-up, and transition of care. Part III of the guidelines will focus on recommendations for lifelong follow-up of ARM, integration of care, and transition of care from pediatric to adult specialists.

Methods

The methods for the adaptation of the Dutch Quality Standard for ARM have been reported previously (part I). Briefly, the Dutch Quality Standard was appraised according to the Appraisal of Guidelines for Research and Evaluation instrument and recommendations were adapted in adherence with the ADAPTE method (detailed methodology available in [supplementary material]).[11] [13] A systematic review of the literature since 2017 was performed according to the PRISMA guidelines and new studies relevant to the lifelong follow-up and transition of care to adulthood of ARM patients were identified in the following databases: Medline, Embase, and Cochrane. The original search strategy as well as inclusion and exclusion criteria for studies are outlined in the [supplementary material]. Original recommendations and newly identified studies were assessed by the AAWG, a panel of 15 experts from 7 European countries, including two patient representatives, within the ERN eUROGEN during a face-to-face meeting in Nijmegen, The Netherlands. Recommendations were either adopted, adapted, or newly developed based on the available evidence ([Table 1]). A grade of recommendation was issued by the AAWG and methodologists (WI, KM) for all recommendations to improve the rigor of development compared with the original quality standard ([Table 2]). For some recommendations, it was unclear from the Dutch Quality Standard whether this recommendation was based on expert opinion only or if there was also supporting evidence. In this case, the recommendation was discussed by the AAWG, and a grade D recommendation (“expert opinion”) was chosen if there was no supporting evidence.

Table 1 Definition of adopted, adapted, and new recommendations

Adopted	Recommendation is endorsed by the AAWG without changes
Adapted	Recommendation was modified and adapted to the needs and context of the ERN
New	Recommendation was created based on the included new evidence or expert opinion

Abbreviations: AAWG, Adoption and Adaptation Working Group; ERN, European Reference Network.

Table 2 Correspondence between grades of recommendations and level of evidence of study

A	Based on consistent level 1 studies
B	Based on consistent level 2 or 3 studies or extrapolations from level 1 studies
C	Based on level 4 studies or extrapolations from level 2 or 3 studies
D	Based on expert opinion or inconclusive/inconsistent studies of any level

Results

The following key and sub-questions were re-assessed by the AAWG:

1. What is the optimal follow-up for people with an ARM at an expertise center?

   • What is the (minimum) frequency of follow-up appointments?

   • Which points of attention/questions should be addressed during a follow-up appointment at different ages?

   • Which actions/investigations must be performed during a follow-up appointment at different ages?

2. What measures/steps need to be taken to ensure a smooth transition of children with ARM from child to adult care?

   • Which care provider is responsible for a smooth transition to adult care?

   • At what point will the adult surgeon/gastroenterologist (if necessary)/urologist/gynecologist be involved in the transition process?

A total of eight new studies addressing follow-up and transition of care of ARM patients were identified and considered by the panel in its recommendations.[14] [15] [16] [17] [18] [19] [20] [21] A detailed summary of all newly included studies for each key and sub-question is provided in the [supplementary material]. Overall, the level of newly found evidence was considered to be low. A total of 18 recommendations were adapted from the Dutch “Quality Standard for Anorectal Malformations,” 6 were adopted, and 6 were developed de novo. Key recommendations of the panel according to the available evidence are summarized in [Fig. 1].

Fig. 1 Recommendations for lifelong follow-up and transition of care for patients with anorectal malformation (ARM).

Recommendations for Lifelong Follow-up and Integrated Care for Patients with ARM

1. What is the optimal follow-up for people with an ARM at an expertise center?

   • What is the (minimum) frequency of follow-up appointments?

   • Which points of attention/questions should be addressed during a follow-up appointment at different ages?

   • Which actions/investigations must be performed during a follow-up appointment at different ages?

A structured lifelong follow-up program involving a multidisciplinary team should be made available to all ARM patients. Children and young adults ought to be examined at expertise centers at crucial time points in their lives such as entering kindergarten/preschool and school, puberty, or transition to adulthood as part of an organized follow-up program ([Table 3]). An example for a structured lifelong follow-up program was outlined in the Dutch Quality Standard and can be found in the [supplementary material]. A pediatric surgeon should be the point person or supervising physician (SP) and can involve other specialists if needed, as somatic as well as psychosocial aspects need to be considered. During follow-up, it is essential to monitor digestive and urogenital tract functions.[10] Urodynamic studies need to be performed in every patient with spinal cord dysraphism and in patients with high suspicion of a neurogenic bladder in accordance with the European Association of Urology guidelines, to determine potential risks to the upper urinary tract and assess function of the detrusor muscle and sphincter muscles.[22] Standardized general or disease-specific questionnaire should be utilized to monitor quality of life and digestive tract function.[14] [23] Subjective scoring systems for bowel function such as Holschneider's or Rintala's questionnaires have not been validated for the purpose of bowel follow-up and do not adequately assess psychosocial issues related to incontinence oftentimes experienced by patients and are therefore considered to be of limited usefulness by the expert panel but may be used as initial screening tools.[6] Additionally, although manometry can provide information about bowel and sphincter function, it is not generally practiced or recommended by the AAWG, as lack of standardized protocols limits the utility and comparability of this invasive procedure.[24] [25] Furthermore, it can lead to significant anxiety in patients, especially young children.[26] Medical providers should also keep in mind the potentially increased risk of colorectal cancer in young adults with ARM. Although the exact risk of colorectal cancer in ARM patients has not yet been established and there is no evidence to support the implementation of specific screening programs, incidental cases have been reported warranting the need for further investigations.[27]

Table 3 Recommendations for lifelong follow-up and integrated care for patients with ARM

Lifelong follow-up and integrated care		Grade of recommendation
1	It is recommended to see all children and young adults at the expertise center for ARM at crucial times of age and an appointment/visit should be offered in an organized follow-up program. [adapted]	D
2	After the ARM has been surgically repaired, the child is guided through a structured lifelong follow-up program. See Table 3.1 in the Dutch Quality Standard.[12] [adapted]	D
3	The organized follow-up program should be performed by a pediatric surgeon (supervising physician). If needed the patient should be offered a multidisciplinary team. [adapted]	D
4	Throughout the follow-up, the function of the digestive tract and the urogenital tract will be monitored. [adapted]	B[14]
5	It is recommended to monitor quality of life during follow-up with standardized general or disease-specific questionnaires. [adapted]	D[23]
6	Scar morbidity and symptoms should be addressed during follow-up. If necessary, a plastic surgeon can be consulted. [new]	C[15]
7	Patients should be given access to information on quality of life and mental health, for example, through: https://eurogen-ern.eu/ [new]	D
8	During puberty, adolescence, and adulthood, attention is paid to the possible psychosocial and sexual problems: information material with regard to sexual function/issues should be provided, for example: https://sexuality-arm-hd.com/, or through patient support groups or experienced professionals. [adapted]	C[16]
9	The parents of the child with ARM receive information not only immediately after birth but also during follow-up. Referral to a patient organization is recommended as these organizations can provide additional information and support that can contribute to improving quality of life. [adapted]	D[17]
10	If patient associations are available, refer patients with ARM to patient associations for support and education about self-management of the malformation. [adapted]	C[32]
11	An experienced nurse/case manager should be involved in the postoperative care and lifelong follow-up. [adapted]	D
12	Nutrition, growth, and neuromuscular development are important considerations during all follow-ups. [adapted]	D
13	(Neuro)psychological follow-up is recommended on indication in children with ARM (poor academic performance or potential shorter attention span, possibly due to physical problems). [adapted]	C[18]
14	Gynecological follow-up is recommended on indication in females with ARM (especially in females with a cloacal malformation or with another known malformation of the gynecological tract), to detect abnormalities/clinical symptoms on time, or to follow up on known anomalies. In case of complaints, an ultrasound of the lower abdomen must be performed during adolescence around the time of the menarche (on average more than a year after the thelarche/breast development). This is to evaluate whether blood is accumulating in the vagina or uterus because it cannot be drained. [adapted]	D
15	Urodynamic studies should be performed in every patient with spinal cord dysraphism as well as in every child with high suspicion of a neurogenic bladder to estimate the risk for the upper urinary tract and to evaluate the function of the detrusor and the sphincter. [new]	D[22]

Abbreviation: ARM, anorectal malformation.

For females with ARM, especially females with cloacal malformation or other known malformation of the reproductive tract, gynecological counseling should be offered around the time of puberty to detect abnormalities and address clinical symptoms in a timely manner. If complaints arise during adolescence, an ultrasound of the lower abdomen around the time of menarche should be performed to assess accumulation of blood in the uterus resulting from drainage issues.

Scar morbidity and related symptoms should be reported systematically, and the Patient and Observer Scar Assessment Scale can be used for this purpose.[28] If necessary, a plastic surgeon may be consulted.

Additionally, proper nutritional intake, somatic growth, and neuromuscular development need to be considered throughout the follow-up. A dietician can be consulted if a risk of malnutrition is identified.

Attention should be paid to mental health and psychosocial well-being of ARM patients and information on the matter should be provided to both patients and parents (or guardians) via official sources (e.g., eUROGEN ERN website) or patient organizations and support groups.

(Neuro)psychological follow-up is recommended for certain indications such as poor academic performance or difficulties in concentrating, as a recent study has shown that ARM patients are more at risk of suffering from developmental or intellectual disability, anxiety, depression, or hyperactivity disorders compared with their peers.[18]

At the onset of puberty and throughout adolescence and adulthood, the possibility of the occurrence of sexual problems and how to address them need to be discussed with and treated by the primary healthcare provider. Additional support can be given by patient associations. Moreover, patient associations can provide education about self-management, which has proven to be effective in treating incontinence, as well as connect patients with one another, thus promoting peer support and mutual knowledge.[29] [30] [31] [32]

Recommendations for Transition of Care for Patients with ARM

• 2. What measures/steps need to be taken to ensure a smooth transition of children with ARM from child to adult care?

  • Which care provider is responsible for a smooth transition to adult care?

  • At what point will the adult surgeon/gastroenterologist (if necessary)/urologist/gynecologist be involved in the transition process?

There is a high prevalence of fecal and urinary incontinence, chronic constipation, and sexual dysfunction among adult ARM patients.[4] [5] Aforementioned morbidities can translate into impaired health-related or gastrointestinal quality of life and psychosocial well-being, highlighting the need for effective and continued care of ARM patients throughout life.[3] [6] [7] [8] Efficient collaboration, referral, and communication between pediatric and adult medical professionals involved in the care of patients with ARM are essential to ensure a smooth transition from childhood to adulthood. The pediatric team, comprised of a nurse practitioner or case manager and a pediatric surgeon, is responsible for this process ([Table 4]). Additionally, specialists, such as psychologists, can be involved if needed. Treatment centers are encouraged to set up a structured program that can be tailored to individual needs and preferences to facilitate this period. Overall, a shared decision-making approach that incorporates the patients' views and wishes should be adopted, as it promotes a sense of ownership and responsibility for their own health, ultimately benefiting the transition process.

Table 4 Recommendations for transition of care for patients with ARM

Transition care		Grade of recommendation
1	The nurse practitioner (case manager) and pediatric surgeon ensure a smooth transition from childhood to adulthood by preparing young people and their parents. The pediatric team is responsible for this process. If necessary, other specialists will be involved such as the psychologist etc. [adapted]	D
2	During the transition process the patient should be involved in all decisions. [new]	D
3	Treatment centers involved with the care for ARM patients are encouraged to install a structured transition program. [new]	D
4	A personalized transition plan should be handed out to all ARM patients at the beginning of the transition process. [new]	D
5	It is recommended to create a tailored transition plan for each individual patient, considering their condition(s), intelligence, and capabilities. [adopted]	D
6	The transition planning typically starts at the age of 12 to 14 years but can differ individually. The actual transition to adult care takes place depending on the local regulations for adulthood in healthcare. [adapted]	D
7	The care-providing expertise centers must take their responsibility for the transition of care of patients with ARM through the pediatric specialists to the adult equivalent (surgeon, urologist, etc.). The ideal transition seems to consist of the involvement of both the pediatric specialist and an involved adult specialist, resulting in a collaboration between the two in which easy contact and consultation are possible. [adopted]	C[20]
8	During the transition phase, all the involved healthcare providers should pay extensive attention to the establishment of a positive transfer to a new doctor in charge of adult care. [adapted]	D[33]
9	Good cooperation and communication between the gastroenterologist/surgeon/urologist/gynecologist, where the adult patient is being treated, and the pediatric surgeon/pediatric gastroenterologist/pediatric urologist, who knows the history, are important. [adopted]	D
10	Follow-up for adults is not yet well organized everywhere, and many adolescents do not come to the clinic. The healthcare professionals and patients should be encouraged to provide/attend follow-up appointments/visits within an organized and structured setting. [adapted]	-D
11	Personal attention, compassion, and support are the most important factors that create a familiar environment that results in the desire of patients with ARM to continue to visit the clinic. [adopted]	D
12	In addition to guidance in the field of ARM and the (possible) medical consequences of the condition, attention is also given to: problems that puberty can bring, the future relationships, sexuality, and fertility. Attention is also paid to the role of the parents during the consultations, as well as the patient's expectations and preferences regarding the transition. [adopted]	D
13	In patients with ARM who have questions or concerns about having children, the threshold for referral to preconception care and consultation should be low. [adapted]	D
14	Good guidance, a transition coordinator, a transition brochure, and combined transition consultations with a child and adult care provider are part of a good protocol for structured preparation for the transition. [adopted]	D
15	The patients must have open access to their data, either through an overview provided by their supervising physician or by requesting a copy of their status. This is to prevent loss of data due to relocation or changes in patient files in the hospital. [adapted]	D

Abbreviation: ARM, anorectal malformation.

Typically, the planning of the transition starts at the onset of puberty around the age of 12 to 14 years, when patients have a better understanding of their disease although this can vary individually. National healthcare system regulations need to be taken into consideration, but the transition should be completed before the age of 18. The care-providing expertise center or pediatric surgery center is responsible for the transition of the patient from the pediatric specialist to the adult equivalent.[20] Ideally, both the parties should be involved at this stage to ensure easy collaboration, communication, and consultation between patients, parents, and medical professionals. Not only expectations of patients but also of parents ought to be considered.

To date, follow-up for adults is unfortunately oftentimes not well organized, leading to a loss of follow-up during adolescence. Healthcare professionals and patients as well as their parents should be encouraged to provide/attend follow-up appointments and a structured setting may be helpful in achieving this goal. Additionally, a positive transfer to the new doctor in charge of adult care should be the goal for all ARM patients.[33] This entails guaranteeing good cooperation and communication between the gastroenterologist/surgeon/urologist/gynecologist and the pediatric equivalent, who is knowledgeable about the case history. It also means understanding past experiences, concerns, and preferences to develop a tailored transition plan and increase patient's satisfaction and quality of life. Additionally, patients should have unrestricted access to their past medical history. An overview can be provided by the SP or patients can request a copy of their chart to prevent loss of data during this critical period.

Lastly, it is known that ARM patients with complex malformations, such as patients with cloacal malformation or bladder neck fistula, can have fertility problems; therefore, the threshold for collaboration or referral to preconception and/or perinatal care specialists should be low.[2] [34] [35]

Evidence Gaps and Future Research

Studies on how the implementation of a structured follow-up program benefits ARM patient care and outcomes are lacking. Similarly, the effectiveness of transition programs with interdisciplinary teams composed of adult and pediatric specialists has not been evaluated to date and objective quality metrics need to be established to evaluate their success. However, considering the high prevalence of persistent symptoms in the adult ARM population, it can be assumed that patients benefit from patient-centric transitional approaches as has been the case for other congenital anomalies.[36] Unawareness of adult physicians about the management of congenital malformations and loss of follow-up during adolescence further impede patient care and highlight the importance of expert centers, a designated SP, and a supporting network provided by patient organizations.

Conclusion

Successful lifelong follow-up and transition of care require a dedicated team of pediatric and adult specialists. The responsibility of transitioning ARM patients to adult care with an individually tailored patient-centered approach lies within the pediatric team. This third part of the eUROGEN guidelines on the management of ARM, developed by an expert panel comprising European pediatric surgeons and patient advocates, summarizes the best available evidence on follow-up and transitional care and provides guidance for the development of structured transition programs.

Conflict of Interest

We would like to disclose that two of the authors of this adaptation report (IDB, CEJS) were also authors of the Dutch Quality Standard. However, it is important to note that neither IDB nor CEJS have any conflict of interest to declare and they did not advocate for any particular viewpoints or positions in relation to the content of the Dutch Quality Standard. No conflicting interests were declared from the other authors.

Acknowledgments

We would like to acknowledge Elena Stallings, Patricia Gavin Benavent, Maria Jose Vicente Edo, and Montserrat Salas Valero from Aragon Health Sciences Institute (IACS, Spain) for their work on this project within the TENDER SANTE/2018/B3/030 “ERN Clinical Practice Guidelines (CPGs) and other Clinical Decision Support Tools (CDSTs).” Further, we would like to acknowledge the representatives of the patient organizations AIMAR and SoMA e.V. for their valuable insights and input.

Collaboration

This work was performed in collaboration with experts involved in ERN ERNICA, EUPSA, and Qualicura.

^* These authors contributed equally.

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Publication History

Received: 14 August 2024

Accepted: 27 August 2024

Article published online:
19 September 2024