## Protocol Section
### Identification Module
**NCT ID:** NCT06424392

**Acronym:** MALFPULM2

**Brief Title:** Respiratory and Psychological Impact of Elective Surgery of Congenital Lung Malformations

**Official Title:** Respiratory and Psychological Impact of Elective Surgery of Congenital Lung Malformations

#### Organization Study ID Info
**ID:** 2024-A00576-41
#### Organization
**Class:** OTHER
**Full Name:** Assistance Publique - Hôpitaux de Paris
### Status Module
#### Completion Date
**Date:** 2026-07

**Type:** ESTIMATED

#### Expanded Access Info
#### Last Update Post Date
**Date:** 2024-05-22

**Type:** ACTUAL

**Last Update Submit Date:** 2024-05-16

**Overall Status:** NOT_YET_RECRUITING

#### Primary Completion Date
**Date:** 2026-07

**Type:** ESTIMATED

#### Start Date
**Date:** 2024-07

**Type:** ESTIMATED

**Status Verified Date:** 2024-05

#### Study First Post Date
**Date:** 2024-05-22
**Type:** ACTUAL
**Study First Submit Date:** 2024-05-16
**Study First Submit QC Date:** 2024-05-16
### Sponsor Collaborators Module

#### Lead Sponsor

**Class:** OTHER

**Name:** Assistance Publique - Hôpitaux de Paris

#### Responsible Party

**Type:** SPONSOR

### Oversight Module

**Is FDA Regulated Device:** False

**Is FDA Regulated Drug:** False

**Oversight Has DMC:** False

### Description Module

**Brief Summary:** The absence of surgery of Congenital lung malformations (CLMs), if it avoids a stressfull event, is accompanied by a "medicalization" of the child, which will be regularly followed up in a specialized medical and surgical environment. The persistent risk of complication, albeit low, is likely to induce over-protective parental behaviours, and to be associated with a sustained family anxiety reaction. The main objective is to test the hypothesis that the absence of surgery has a significant impact on parental anxiety, measurable at 6- 9 years of age.

**Detailed Description:** The knowledge of CLMs has been revolutionized by prenatal imaging and the identification of large numbers of CLMs that remain asymptomatic. France is a leader in this field, having set up the only prospective multicentre cohort currently available internationally, with follow-up starting in the prenatal period (MALFPULM). This cohort has already enabled a better description of the prenatal history and the development of an algorithm predictive of the risk of neonatal respiratory distress. The children were followed up to the age of 2 years, and 66% of them were operated on between 0 and 2 years of age. This cohort is a unique opportunity to measure the mid-term impact of this surgical decision, in terms of both medical complications and psychological consequences.

In particular, the size of the cohort makes it possible to answer with a sufficient level of evidence to the following controversies:

* What is the risk of CLM infection in the absence of surgical removal, and is this risk dependent on the CLM phenotype?
* What is the functional respiratory impact of surgical techniques (thoracoscopy or thoracotomy), depending on the age of the surgery?
* What is the prevalence of musculoskeletal complications according to surgical techniques (thoracoscopy or thoracotomy)?
* What is the burden of the medical or surgical follow-up depending on the chosen therapeutic option?
* What is the psychological impact of the surgical decision on the parents and the child? Considering the psychological impact is a major issue for this malformative condition which mainly concerns asymptomatic children, and is of great originality because it has never been evaluated. The most recent literature clearly calls for integrating issues of family well-being and parental mental health into the follow-up of children with chronic disease and/or congenital anomalies. For this reason, the investigators chose maternal anxiety as the main criterion of this study. Specifically for CLM, demonstrating the impact of the investigator's decisions on the psychological state of the parents will be a strong encouragement to integrate this dimension in care, for a better detection of these anxious and/or depressive parental reactions, and a better personalization of the transmission of decisions.

### Conditions Module

**Conditions:**

- Congenital Lung Malformations

**Keywords:**

- Congenital lung malformations

- Psychological impact

### Design Module

#### Design Info

**Observational Model:** COHORT

**Time Perspective:** PROSPECTIVE

#### Enrollment Info

**Count:** 434

**Type:** ESTIMATED

**Study Type:** OBSERVATIONAL

### Arms Interventions Module

#### Arm Group 1

**Description:** 6-9 years children with fetal diagnoses of CLM and included in MALFPULM who have had at least one follow-up visit between 0 and 2 years of age.

**Intervention Names:**

- Other: General Health Questionnaire (GHQ-28)

- Other: State-Trait Anxiety Inventory (STAI-Y)

- Other: Revised Children's Manifest Anxiety Scale (RCMAS)

- Other: World Health Organization Quality of Life (WHOQOL-BREF)

- Other: Quality of life measure for children (KIDSCREEN-27)

- Other: Parental Educational Competence Self-Evaluation Questionnaire "Questionnaire d'Auto-Évaluation de la Compétence Éducative Parentale (QAECEP)"

- Other: Parental interview

**Label:** 6-9 years children with fetal diagnoses of CLM

### Interventions

#### Intervention 1

**Arm Group Labels:**

- 6-9 years children with fetal diagnoses of CLM

**Description:** Anxiety and depression self-reported scales for both parents

**Name:** General Health Questionnaire (GHQ-28)

**Type:** OTHER

#### Intervention 2

**Arm Group Labels:**

- 6-9 years children with fetal diagnoses of CLM

**Description:** Anxiety and depression self-reported scales for both parents

**Name:** State-Trait Anxiety Inventory (STAI-Y)

**Type:** OTHER

#### Intervention 3

**Arm Group Labels:**

- 6-9 years children with fetal diagnoses of CLM

**Description:** Anxiety and depression self-reported scales for children

**Name:** Revised Children's Manifest Anxiety Scale (RCMAS)

**Type:** OTHER

#### Intervention 4

**Arm Group Labels:**

- 6-9 years children with fetal diagnoses of CLM

**Description:** Quality of life self-reported scales for parents

**Name:** World Health Organization Quality of Life (WHOQOL-BREF)

**Type:** OTHER

#### Intervention 5

**Arm Group Labels:**

- 6-9 years children with fetal diagnoses of CLM

**Description:** Quality of life self-reported scales for children

**Name:** Quality of life measure for children (KIDSCREEN-27)

**Type:** OTHER

#### Intervention 6

**Arm Group Labels:**

- 6-9 years children with fetal diagnoses of CLM

**Description:** Parental Educational Competence Self-Evaluation self-reported Questionnaire for parents

**Name:** Parental Educational Competence Self-Evaluation Questionnaire "Questionnaire d'Auto-Évaluation de la Compétence Éducative Parentale (QAECEP)"

**Type:** OTHER

#### Intervention 7

**Arm Group Labels:**

- 6-9 years children with fetal diagnoses of CLM

**Description:** The interview will only last about ten minutes and will take place during the first call, if the parents have agreed and are sufficiently available to answer. If the parents are not available, an appointment can be made at a later date

**Name:** Parental interview

**Type:** OTHER

### Outcomes Module

#### Primary Outcomes

**Description:** maternal anxiety level measured with the GHQ-28 anxiety scale

**Measure:** maternal anxiety level

**Time Frame:** 1 year

#### Secondary Outcomes

**Description:** Number of lower respiratory tract infections, either documented (radiological opacity) or not, in the last 12 months and from birth to the date of evaluation

**Measure:** Impact of the surgical decision on respiratory morbidity - lower respiratory tract infections

**Time Frame:** 1 year

**Description:** Number of hospitalizations for respiratory reasons in the last 12 months and from birth to the date of evaluation

**Measure:** Impact of the surgical decision on respiratory morbidity - hospitalizations

**Time Frame:** 1 year

**Description:** Number of severe wheezing respiratory exacerbations in the last 12 months, defined as requiring oral corticosteroids, emergency visit and/or hospitalization

**Measure:** Impact of the surgical decision on respiratory morbidity - severe wheezing respiratory exacerbations

**Time Frame:** 1 year

**Description:** Current regular treatment for respiratory purposes, defined as daily treatment for at least 3 consecutive months in the last 12 months

**Measure:** Impact of the surgical decision on respiratory morbidity - treatment

**Time Frame:** 1 year

**Description:** Following parameters will be collected: date of performance, weight and height at date of performance, total lung capacity (TLC), forced vital capacity (FVC), forced expiratory volume in one second (FEV1), Forced expiratory flow at 25-75% (FEF25-75%), transfer factor of the lung for carbon monoxide (TLCO). FVC, FEV1, and FEF25-75%. These measurements will allow to identify following functional profiles:

* lung function restriction, defined by TLC lower than -1.645 Z score
* airway obstruction, defined by FEV1/FVC lower than -1.645 Z score, and its reversibility, defined by an improvement of at least 12% of the FV1 after inhalation of salbutamol.
* impaired diffusion capacity, defined by DLCO lower than -1.645 Z score.

**Measure:** Impact of the surgical decision on respiratory morbidity - lung function tests

**Time Frame:** 1 year

**Description:** The parents's declare rate of thoracic deformities

**Measure:** Impact of the surgical decision on respiratory morbidity - thoracic deformities

**Time Frame:** 1 year

**Description:** The burden of the medical or surgical follow-up will be evaluated by the number of specialist consultations and the number of CT or MRI scans

**Measure:** Impact of the surgical decision on respiratory morbidity - burden of the medical or surgical follow-up

**Time Frame:** 1 year

**Description:** The 28-item GHQ (General Health Questionnaire) was designed to be a self-administered screening test aimed at detecting minor psychiatric disorders and had been and validated in different languages, including French (34). It allows to estimate the prevalence of psychological distress in a given population. The GHQ-28 has been divided into four subscales. These are: somatic symptoms (items 1-7); anxiety/insomnia (items 8-14); social dysfunction (items 15-21), and severe depression (items 22-28). It is scored from 0 to 3 for each response with a total possible score on the ranging from 0 to 84. Using this method, a total score of 23/24 is the threshold for the presence of clinically important psychological distress.

**Measure:** Anxiety and depression - GHQ-28

**Time Frame:** 1 year

**Description:** The State-Trait Anxiety Inventory (STAI-Y) is a 20-item self-measure of state anxiety level in parents, complementary of GHQ-28. It reflects the current subjective feeling of tension, apprehension, nervousness, and worry and is widely used both in practice and in clinical research.

**Measure:** Anxiety and depression - STAI-Y

**Time Frame:** 1 year

**Description:** The RCMAS is a 37-item, self-report instrument designed to assess the level and nature of anxiety. A Total Anxiety score is computed based on 28 items, which are divided into three anxiety subscales: physiological anxiety (10 items about somatic manifestations of anxiety such as sleep difficulties, nausea and fatigue), worry/oversensitivity (11 items measuring obsessive concerns about a variety of things, most of which are typically vague and ill-defined, as well as fears about being hurt or emotionally isolated), and social concerns/concentration (7 items measuring distracting thoughts and fears that have a social or interpersonal nature). The remaining nine items on the RCMAS constitute the Lie subscale. A high score indicates a high level of anxiety or lie on that subscale.

**Measure:** Anxiety and depression - RCMAS

**Time Frame:** 1 year

**Description:** The WHOQOL-BREF is an abbreviated form of the WHOQOL-100 with only 26 items. It is an instrument for recording subjective quality of life. This is defined as an individual perception of one's own life situation in the context of culture and value system as well as personal goals, expectations, evaluation criteria and interests. It is a self-questionnaire that includes the four domains "physical health", "psychological well-being", "social relationships" and "environment". The items are answered using a five-level scale. The scales have a very high internal consistency. It is validated in several languages, including French.

**Measure:** Quality of life - WHOQOL-BREF

**Time Frame:** 1 year

**Description:** The questionnaire KIDSCREEN is used to assess the health-related quality of life of children and adolescents. It was constructed, tested, and normed using data from 3,000 children and adolescents from Europe. The KIDSCREEN-27 was developed to create a shorter version of the KIDSCREEN-52 with a minimum of loss of information and with good psychometric properties. The KIDSCREEN-27 measures the following five dimensions: physical well-being (5 items) ; psychological well-being (7 items) ; autonomy and parent relations (7 items) ; peers and social support (4 items) ; school environment (4 items).

**Measure:** Quality of life - KIDSCREEN

**Time Frame:** 1 year

**Description:** This questionnaire evaluates the parent's sense of competence in his or her role as an educator and has two components: the "skill/knowledge" factor, which evaluates the respondents' perception of the skills and knowledge they have acquired in order to be adequate parents (8 items); the "value/ease" factor, which evaluates the value that the respondent places on the role of parent, as well as his or her ease in this role (9 items).

**Measure:** Parenting Sense of Competence questionnaire

**Time Frame:** 1 year

**Description:** The over-protective parental behaviours is defined by the age of attendance in first community (5 or more children), the care arrangements between 0 and 2 years, the after-school activities in the previous year and the travel outside France in the last two years

**Measure:** Over-protective parental behaviours

**Time Frame:** 1 year

### Eligibility Module

**Eligibility Criteria:** Inclusion Criteria:

* Child of the MALFPULM cohort, with their parents
* At least one follow-up visit between 0 and 2 years of age (n= 414 eligible children)
* Non-opposition of the family

Exclusion Criteria:

* Child with CLM, but not included in MALFPULM
* Parents who participated in MALPULM, but with prenatal fetal death, or neonatal death.

**Maximum Age:** 9 Years

**Minimum Age:** 6 Years

**Sampling Method:** NON_PROBABILITY_SAMPLE

**Sex:** ALL

**Standard Ages:**

- CHILD

**Study Population:** The families of all children included in MALFPULM who have had at least one follow-up visit between 0 and 2 years of age.

### Contacts Locations Module

#### Central Contacts

**Contact 1:**

**Email:** christophe.delacourt@aphp.fr

**Name:** Christophe DELACOURT, MD, PhD

**Phone:** +33 144494838

**Role:** CONTACT

**Contact 2:**

**Email:** laure.choupeaux@aphp.fr

**Name:** Laure CHOUPEAUX, Msc

**Phone:** +33 144381711

**Role:** CONTACT

#### Overall Officials

**Official 1:**

**Affiliation:** Assistance Publique - Hôpitaux de Paris

**Name:** Christophe DELACOURT

**Role:** PRINCIPAL_INVESTIGATOR

### IPD Sharing Statement Module

**IPD Sharing:** NO

## Derived Section

### Condition Browse Module - Browse Branches

- Abbrev: BC16

- Name: Diseases and Abnormalities at or Before Birth

- Abbrev: All

- Name: All Conditions

### Condition Browse Module - Browse Leaves

- ID: M12

- Name: Congenital Abnormalities

- Relevance: HIGH

- As Found: Malformations

### Condition Browse Module - Meshes

- ID: D000000013

- Term: Congenital Abnormalities

### Misc Info Module

- Version Holder: 2024-05-31
