literature review on lean management

A Literature Review on Lean Manufacturing in Small Manufacturing Companies

• First Online: 13 January 2018

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• Laís Ghizoni Pereira 3 &
• Guilherme Luz Tortorella 3  

Part of the book series: Management and Industrial Engineering ((MINEN))

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This chapter aims to identify, through a systematic literature review, the main Lean Manufacturing (LM) practices, critical success factors (CSF) and barriers within small manufacturing companies’ context. This paper presents a systematic literature review based on the proposed approach denoted as ProKnow-C to identify the correlated bibliographic portfolio (BP). Our findings indicate that the consolidation of specific CSF related to the context of small manufacturing companies reinforces the body of knowledge, reinforcing the establishment of a broader perspective of LM implementation in these companies. Further, the capability of disseminating the continuous improvement mindset across all employees is a significant challenge for these companies, since their leaders are poorly trained in accordance with the underlying LM principles. The recent growth of small companies and their relevance to socioeconomic development has raised the importance of improving their management processes. Particularly for LM implementation, few studies have specifically approached this context whose challenges may be differentiated, highlighting the need for a better comprehension of proper practices, barriers, and CSF.

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Appendix—Bibliographical Portfolio

Antony, J., Kumar, M., & Madu, C. N. (2005). Six sigma in small-and medium-sized UK manufacturing enterprises: Some empirical observations. International Journal of Quality & Reliability Management, 22 (8), 860–874.

Belhadi, A., & Touriki, F. (2016). A framework for effective implementation of lean production in small and medium-sized enterprises. Journal of Industrial Engineering and Management, 9 (3), 786–810.

Bhamu, J., & Singh Sangwan, K. (2014). Lean manufacturing: Literature review and research issues. International Journal of Operations & Production Management, 34 (7), 876–940.

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Bhasin, S. (2012). Prominent obstacles to lean. International Journal of Productivity and Performance Management, 61 (4), 403–425.

Doolen, T., & Hacker, M. (2005). A review of lean assessment in organizations: An exploratory study of lean practices by electronics manufacturers. Journal of Manufacturing Systems, 24 (1), 55–67.

Hallgren, M., & Olhager, J. (2009). Lean and agile manufacturing: External and internal drivers and performance outcomes. International Journal of Operations & Production Management, 29 (10), 976–999.

Kumar, M., & Antony, J. (2009). Multiple case-study analysis of quality management practices within UK six sigma and non-six sigma manufacturing small-and medium-sized enterprises. Proceedings of the Institution of Mechanical Engineers, Part B: Journal of Engineering Manufacture, 223 (7), 925–934.

Kumar, M., Antony, J., Singh, R., Tiwari, M., & Perry, D. (2006). Implementing the Lean Sigma framework in an Indian SME: A case study. Production Planning and Control, 17 (4), 407–423.

Manville, G., Greatbanks, R., Krishnasamy, R., & Parker, D. W. (2012). Critical success factors for lean six sigma programmes: A view from middle management. International Journal of Quality & Reliability Management, 29 (1), 7–20.

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Sánchez, M., & Pérez, M. (2001). Lean indicators and manufacturing strategies. International Journal of Operations & Production Management, 21 (11), 1433–1452.

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Shah, R., & Ward, P. (2007). Defining and developing measures of lean production. Journal of Operations Management, 25 (4), 785–805.

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Pereira, L.G., Tortorella, G.L. (2018). A Literature Review on Lean Manufacturing in Small Manufacturing Companies. In: Davim, J. (eds) Progress in Lean Manufacturing. Management and Industrial Engineering. Springer, Cham. https://doi.org/10.1007/978-3-319-73648-8_3

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Lean six sigma in the healthcare sector: A systematic literature review

Rajeev rathi.

a School of Mechanical Engineering, Lovely Professional University, Phagwara 144001, India

Ammar Vakharia

b School of Electrical and Electronics Engineering, Lovely Professional University, Phagwara 144001, India

Mohd Shadab

Healthcare is a very important sector as our lives depend on it. During the novel corona virus pandemic, it was evident that our healthcare organizations still lack in terms of efficiency and productivity. Especially in the developing nations, the problems were much bigger. Lean Six Sigma (LSS) is a methodology which when implemented in an organization, helps to increase the process capability and the efficiency, by reducing the defects and wastes. The present study systematically reviews the research studies conducted on LSS in the healthcare sector. It was found that comparatively less studies are focused on improving the medical processes, most of the studies targeted the management processes. Moreover, lesser number of studies were being conducted for developing nations, but now it seems that the focus of research scholars has shifted towards the developing nations also. But it was observed that the studies in these nations were majorly empirical in nature, very few studies were conceptual or exploratory. There is a need for guiding healthcare professionals on creating a continuous improvement environment, which sustains the improvements achieved after LSS implementation.

1. Introduction

There is little room for error when it comes to healthcare. Simple mistakes can impact hundreds of people and can lead to fatalities. According to the journal of healthcare finance, medical errors cost the US over nineteen point five billion dollars in 2008. The Institute of Medicine in 1998 estimated that 98 thousand deaths could have been prevented that year due to medical errors. While in 2008 that figure had ballooned to 200 thousand deaths a year, that’s five hundred and forty-eight deaths a day or 1 death almost every two minutes in the US alone [1] . Lean Six Sigma can go in a long way in reducing these devastating preventable deaths. A healthcare practitioner who understands how to use the tools and methodology to systematically resolve problems and improve the quality of care is well-positioned to become a highly valued asset to their organization [2] . They not only prevent deaths but also improve the quality of patient care and reduce operating costs. There are plenty of opportunities for improvement in healthcare. Lean Six Sigma combines the strategies of Lean and Six sigma. Lean is famous for its ability to handle waste and Six sigma is known for process improvement [3] . It is well known that six sigma stands for standard deviation. Therefore, to improve the efficiency and quality of the process combining these philosophies will eliminate waste and reduce variability [4] . First lean methodology is used to eliminates the waste then through six sigma tools we can improve process variation so these two methods go hand in hand in today’s time [5] . Combination of lean and six sigma also gives good results in improving the process flow [6] . Principles of six sigma and lean have a lot of similarities, and that’s why lean and six sigma has been practiced together. Both of them, at the end of the day deliver the same kind of value to the customer and to the businesses [7] . We know that lean and six sigma both sort of originated out of manufacturing, a lot of it from manufacturing of automotive. But today it’s used in the public sector, in customer service, in healthcare and can be used everywhere. Lean thinking goes back a long way. Henry Ford kind of established it early on the first mass production system by combining standard parts conveyors and workflow. Later on, Kiichiro Toyota implemented new concepts, like they came up with things like value stream and Kanban, which become to known as Toyota production system in the 90 s [8] . It became more extensively recognized as a solution that works. Lean in manufacturing was introduced by James Womack who works at the lean institute. When taking a lean approach, the general focus is on the qualitative tools. This is one of the reasons that most lean six sigma activity begin with lean, as qualitative tools are little more intuitive. The concept of lean and six sigma is a little easier to understand then to implement [9] .

Day to day rising in cost of healthcare and decrease in reimbursement rates what can hospitals do to ensure operational efficiencies, quality patient safety, and employee engagement while still meeting the bottom line. Lean and Six Sigma have a combine relationship with each other in improving the quality of services by reducing costs and wastes and gives a good result in healthcare improvement [10] . Lean six sigma is not just a methodology or not just having tools that are needed to make an improvement but it is having mentality and psychology to make change happen. As healthcare is people intensive and process driven industry so this is the perfect environment for lean and six sigma. Worldwide, LSS is being implemented in various service as well as manufacturing organisations. Still, healthcare sector is not much familiar with the sustainable benefits of LSS strategy, especially in developing nation like India, Pakistan, Sri Lanka etc. This study has presented a systematic literature review of LSS in developing nations healthcare organisations and the opportunities was explored. With this study, the healthcare professionals will be able to focus on grey areas to effectively improve their organization performance.

2. Literature review

Exploring the literature for a topic or a field is a very essential process. As soon as a person starts with reviewing the literature, a picture forms slowly with each step ahead. It represents all the major contributions for developing the field. Knowledge of various techniques, scientific methodologies and new technologies is gained as a result of the literature review [11] . Further, it navigates towards the direction where more research work and study are required, in order to achieve advancements in the sector. There are three steps in the research methodology for the current study. In the first step, the SLR is conducted. It includes three sub steps of accessing the articles from various sources, then excluding the articles according to the exclusion criteria of SLR and then finally categorizing them according to various parameters. In the second step, the articles are analysed and some of the trends are observed, like which department of a healthcare organization is given an utmost importance in research perspective. Lastly, the future implications are noted, which can act as a useful guide for healthcare professionals to look for in the upcoming time.

2.1. Systematic literature review

Systematic Literature Review (SLR) on the other hand, is different from the traditional literature review method. It is an exhaustive process, where the literature is explored in a systematic manner, as represented in Fig. 1 . It was first introduced by Tranfield, Denyer, & Smart in their research paper published in the year 2003 [12] . SLR is carried out in 3 phases. The first phase deals with planning of the review, while in the second phase the review is conducted after defining few inclusion criteria. Finally, the articles are reviewed in the third phase [13] .To search for the articles, there is a criterion which is mainly focused on the topic related to lean six sigma methodology implication, LSS framework in healthcare and healthcare sector itself to include/exclude the articles. Moreover, the language of the articles is also considered for the inclusion/extrusion of the articles. In spite of this formal search and collection strategy, was needed to ensure the thoroughness of the literature review which is mainly focused on the topic (healthcare sector). As a result, articles that simply did not discuss the issue of lean six sigma in the healthcare sector were excluded. So, for the current study, hundred and forty articles were analysed but some of them were extruded due to the following barriers:

• • Language of the article (means articles must be only in English).
• • Article must have the concept of lean six sigma.
• • Article must be on healthcare sector.

Systematic Literature Review.

The research articles can be identified according to three main characteristics, namely place, research category and time, as shown in Fig. 2 . Place depicts the country where the research study is focused on. A country can be a developed nation, or a developing nation. While the research category means the type of article or the method used in the article. Research category can be categorized further into 4 types, namely conceptual, descriptive, empirical and exploratory. A conceptual research represents theories and ideas, which are developed using observations. A scholar may introduce a new concept or upgrade an existing methodology, but no experimentation work is generally required here [14] . Whereas a descriptive research study is an inclusive research work which represents all the major contributions, changes or things happening in relation to the field under scope of the project [15] . In order to collect data, surveys are conducted which makes it's obvious that the whole study is navigated solely by the data received and the research scholars have no control over it. Empirical research study is more of an experimental study, which uses observational methods to collect data and then experimentation is done to verify that observations [16] . While an exploratory study tries to address a new topic or a problem whose solution is not yet obtained or is at preliminary stage of development. Exploratory study helps in designing methods which may lead towards a solution or help better understand the field [17] . The time indicates the year in which the study was conducted or published.

Characteristics of research papers.

2.2. Systematic literature review of LSS in healthcare

Healthcare is categorized as a service sector, where service in the form of medical aid is provided. It is very important to rigorously upgrade the healthcare system, in terms of efficiency and productivity. As the healthcare is not just merely a business, but a system which has an impact on all lives. After the introduction of Six Sigma (SS) methodology in 1986 by Bill Smith, and Lean methodology in 1988 by John Krafcik, it took nearly a decade for these individual methodologies to be introduced in the healthcare sector [18] . Very few research studies were conducted for the Lean and SS in healthcare during the early days. In the year 2001, Lean and Six Sigma integration was in the trend. Yet, the use of LSS methodology in healthcare was not seen before the year 2005 [19] . The integration of Lean with Six Sigma results in a method which can reduce wastes and improve productivity, as well as reduce errors and improve patient satisfaction level [20] .A total of 80 articles are considered for the present study. Table 1 lists 47 articles which addresses the developed nations, and the articles which were not written specifically for any nation. While Table 2 contains 33 articles whose main focus was on developing nations, and the healthcare organizations in that countries.

LSS in Healthcare: Developed Nations and Global.

2. LSS in Healthcare: Developing Nations.

Empirical studies were conducted while focusing on a particular hospital or a particular department in a hospital, like Usha Manjunath et. al. 2007 [64] implemented LSS methodology in an Indian hospital, while Giovanni Improta et al 2019 [59] implemented LSS to reduce the length of stay of patients in an Italian hospital, as listed in the table 1 . Research studies focusing on reviewing the literature plays a crucial part in the development of a field, because it shows the complete picture which tells about the overall advancements and the areas where still more study is required. Similarly, for the healthcare sector studies have been conducted to review the literature available. For an example, Peimbert-García et al 2019 [ 55 ] conducted a review and directed the research scholars towards the areas in healthcare which required more focus. Whereas, the scholars have also focused on topics which makes the implementation of LSS in healthcare much easier. Like, V. Vaishnavi et al 2020 [ 90 ] identified 16 readiness factors for LSS implementation in healthcare. These factors should be fulfilled by a healthcare organization before starting with the implementation phase of LSS [96] .Certain crucial factors which affect the implementation of LSS are -:

• • Management willingness and effective leadership
• • Resource capabilities
• • Employee training and trust
• • Financial capabilities
• • Availability of expertise and knowledge

3. Results and discussions

LSS implementation in the healthcare began not earlier than 2005. A lot of research has already been done on LSS in healthcare all over the world, but when it comes to developing nations like India and Brazil, comparatively less research has been conducted [97] .The time constraint trend in catagorisation of the articles, is crucial to indicates how the trends is changing over the year and how many researches are conducted in a year to overcome the problem. Moreover, the time also indicates the change of area of focus of the researchers with changing time. Until the year 2013, research scholars focused majorly on improving the healthcare sector of developed nations, as evident from Fig. 3 .However, commencing from 2014 the focus shifted to developing nations, as they are the ones which require more efficient systems.41 percent of the studies targeted the organizations in developing nations, and further 30 percent of articles from the developing nations category addressed the Indian healthcare organizations, as shown in Fig. 4 .It is important to note this trend because a study related to a particular place demonstrates the importance and need of the study for a unique problem and its solution related to that place. Because as the places differ problems also varies. So that it could be defined as per the requirement related to that particular place.

Year wise distribution of the research articles.

Categorization of the articles according to place.

Fig. 5 helps to distribute the articles according to the type of process in the healthcare organization they target, namely medical processes, management processes and general processes. Further, the articles are also divided according to the place they have focused on. It is evident from the Fig. 5 that comparatively less articles have addressed the medical processes. Most of the research studies conducted in the developing nations were empirical, as depicted in Fig. 6 . Whereas, there is a gap when it comes to exploratory studies and conceptual studies. This trend is significant because, if in a particular country there are less exploratory studies carried out, than the problems unique to that particular country won’t be decomposed. So, there must be an exploratory study to demonstrate the simple and feasible solution. The researchers need to devise new concepts and address the problems which are still not solved yet, while targeting not just one organization but the whole nation. For an example, Sreeranga Bhatt et al 2014 [73] implemented the LSS methodology using DMAIC in a hospital, similarly some other researchers have successfully implemented LSS in one or in some cases up to five hospitals. But this successful research work method cannot be applied to all the hospitals, as all have their own unique problems.

Characteristics of articles according to process.

Research category in developing nations.

3.1. Implications and future opportunities

After analysing the articles included for the current study, some areas or topics were found which lacked in terms of research. Moreover, these can be taken as future opportunities for the research scholar and the healthcare professionals to work on. Healthcare practitioners may be able to identify fields that need more study and, as a result, better strengthen their organizations. The following are the topics which will set direction for future research:

• 1. Medical process: More study is needed to apply the LSS approach in order to minimize defects and wastes associated with surgical procedures such as therapies and operating rooms. The majority of studies were found to be either for managerial processes or were conducted aiming the whole organizational structure.
• 2. Continuous improvement culture: After the implementation of the LSS approach, healthcare managers must be motivated in developing a continuous improvement philosophy in order to sustain the gains made and identify potential prospects. As per the research done by the authors, no studies were found to address this topic.
• 3. Sustainability: It is important to understand environmental factors as well, as the healthcare industry generates bio hazardous wastes that harm the ecosystem. LSS can be integrated with green technologies to have a sustainable improvement framework.
• 4. Supply chain: As evident during the pandemic, the supply chain of the healthcare sector was not quite effective and ready for these kinds of uncertain situations. But the bad times always gives a lesson, and hence it is the best time to reflect on the problems and build an LSS framework which can help even during pandemic.
• 5. More research work is required in developing nations. Moreover, most of the studies were conducted in India, very less aimed at other developing nations like Sri Lanka, Pakistan, Bhutan, and Bangladesh.
• 6. Most of the studies in the developing nations were empirical in nature, more conceptual and exploratory are required. Because these types of studies have the potential to address the unique problems for each individual country. As each country has their own policies and rules, which could produce a challenge for a LSS model which was successful in other nation.

There is a need for improving the performance of healthcare organizations. These organizations faced a lot of difficulty during the corona virus pandemic. Therefore, to avoid such situations in future it has become very critical for the healthcare managers to implement the process improvement methodologies like Lean Six Sigma. Moreover, the managers and their staff are motivated to implement LSS to reduce the defects and wastes related to medical processes like during the surgeries and in operation theatres. After the initiation of LSS methodology, the healthcare managers need to create a continuous improvement culture in their organization, so as to maintain the improvements done and find new opportunities. Lastly, the healthcare professionals and managers need to consider the environmental aspects too, as healthcare sector has bio hazardous wastes which has damaging effects on environment. Lean Six Sigma can be combined with green methodology in order to deal with this [98] . The integrated green lean six sigma could become an effective booster to enhance the quality, effectiveness and efficiency of a particular organisation. Therefore, the management of the healthcare organizations are motivated to implement this method to develop their organization in a sustainable manner [99] . Yet, it is evident that still the journey for LSS implementation in the healthcare sector is a long way ahead, especially for the developing nations.

4. Conclusion

Healthcare is a very crucial and complex sector, it involves several departments, and a failure in one department may have a negative impact on patient's care. It was also evident during the pandemic due to the Covid-19, that we need an operational excellence in healthcare operations. Lean Six Sigma is a methodology which can reduce wastes and variation in an organization with the help of lean and six sigma tool set. Lean Six Sigma is a well-established methodology, mostly used in the manufacturing sector [100] . However, in the past decade it is being extensively used for the non-manufacturing industry also, like the service sector. There has been a shift in number of studies being conducted according to place. The researchers have acknowledged the gap, when it comes to developing nations, and the number of studies conducted has increased for these nations [101] . Present study shows that till, in healthcare sector, LSS is mainly limited in management of operations, and more research is needed in eliminating waste and defects in surgical and operation theatre activities. Furthermore, guidance should be provided to the healthcare professionals on how to create a continuous improvement culture, and then sustain the environment and the improved processes. Researchers are working tirelessly in order to make the healthcare organizations perfect and efficient, so that everyone's life can be improved. Indeed, it is the healthcare sector on which we are dependant for life support.

5. Limitations

An exhaustive systematic literature review was conducted for the present study. The major limitation of this study is that during the exhaustive literature review a number of articles were filtered out which are in language other than English, not related to the healthcare sector, not using the LSS methodology, and not yet published. As a result, a very clear scenario of LSS in the healthcare sector might not be presented. Secondly, the study has not included a LSS framework which can guide the healthcare professionals to implement the LSS methodology in their organizations, but this can be a part in a consecutive paper.

CRediT authorship contribution statement

Rajeev Rathi: Supervision, Conceptualization, Resources, Writing - review & editing. Ammar Vakharia: Conceptualization, Formal analysis, Investigation, Visualization, Validation, Writing - original draft. Mohd. Shadab: Data curation, Formal analysis, Investigation, Visualization, Validation, Writing - original draft.

Declaration of Competing Interest

The authors declare that they have no known competing financial interests or personal relationships that could have appeared to influence the work reported in this paper.

Lean Management Systems in Health Care: A Review of the Literature

Affiliation.

• 1 Johns Hopkins University School of Nursing, University in Baltimore, Maryland (Dr Winner); Lean Sigma Deployment, Armstrong Institute for Patient Safety and Quality, Johns Hopkins University School of Medicine, Baltimore, Maryland (Dr Winner and Ms Reinhardt); Armstrong Institute for Patient Safety and Quality, Johns Hopkins University School of Medicine, Baltimore, Maryland (Dr Benishek); and Center for Health Services and Outcomes Research, Johns Hopkins Bloomberg School of Public Health, Baltimore, Maryland (Dr Marsteller).
• PMID: 35180733
• DOI: 10.1097/QMH.0000000000000353

Background and objectives: Many health care organizations now employ Lean tools to improve value in health care, yet reports of their effectiveness vary. This variation may be explained by the context in which Lean is implemented, whether as a tool or as a management system. This article reports on a structured literature review conducted to understand the evidence base for the impact of Lean Management System implementation in health care.

Methods: A search of PubMed, Scopus, Emerald, EMBASE, CINAHL, and Business Source Complete databases was conducted in November 2017 and repeated in July 2020 to assess the evidence for the impact of Lean Management Systems in health care from 2000 to July 2020. Articles were included if they (1) reported on a Lean Management System or (2) reported on Lean Management System components as described by Mann 1 (ie, leader standard work, visual controls, daily accountability process, and discipline).

Results: A total of 52 articles met the inclusion criteria. Although all articles described some combination of leader standard work, visual management, and daily accountability as part of their Lean Management System, only a handful described use of all 3 components together. Only one explicitly mentioned the fourth component, discipline, required to consistently apply the first 3. The majority reported on single-unit or department implementations and most described daily huddles at the unit level that included review of key performance indicators, identification of improvement opportunities, and problem solving. The role of the leader in a Lean Management System was described a coach and a mentor. Barriers to adoption such as insufficient training and increased workload for nurses were noted along with the importance of relevance to the local context for unit teams to find value in huddle boards and huddles. As yet, evidence of Lean Management System effectiveness in driving health care improvement is absent due to weak study designs and lack of statistical rigor.

Conclusion: Well-designed research on Lean Management Systems in health care is lacking. Despite increasing adoption of Lean Management Systems over the past 10 years and anecdotal reports of its effectiveness, very few articles provide quantitative data. Those that do report unit-level implementation only, little use of a comprehensive package of Lean Management Systems elements, and weaker study designs and statistical methods. More rigorous study designs and robust statistical analysis are needed to evaluate effectiveness of Lean Management Systems in health care. This represents a rich area for future health care management research.

Copyright © 2022 Wolters Kluwer Health, Inc. All rights reserved.

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• Delivery of Health Care
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• Problem Solving

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Please note you do not have access to teaching notes, lean management practices in healthcare sector: a literature review.

Benchmarking: An International Journal

ISSN : 1463-5771

Article publication date: 22 January 2019

Issue publication date: 25 April 2019

The purpose of this paper is to synthesise the extent to which lean implementation in healthcare has been studied in the literature since its inception a decade ago.

Design/methodology/approach

This paper is based upon a literature review of mostly academic articles published mainly in the fields of operations management and medicine.

The current state of the literature on lean healthcare implementation is primarily evaluative (benefits-oriented), descriptive (process-oriented) and rarely holistic (interaction of lean implementation and clinical practice).

Originality/value

This paper identifies further research directions for academics, and provides an overview of findings relevant to healthcare stakeholders interested in lean implementation.

• Implementation

Acknowledgements

The author would like to thank all those who are a part of my research journey.

Parkhi, S.S. (2019), "Lean management practices in healthcare sector: a literature review", Benchmarking: An International Journal , Vol. 26 No. 4, pp. 1275-1289. https://doi.org/10.1108/BIJ-06-2018-0166

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Case report

• Open access
• Published: 21 April 2024

Peripartal management of dichorial twin pregnancy in a bicornuate bicollis uterus: a case report and review of the literature

• Melanie Schubert   ORCID: orcid.org/0000-0002-2806-778X 1 ,
• Anastasia Tihon 2 ,
• Kristin Andresen 1 ,
• Zino Ruchay 1 ,
• André Farrokh 1 ,
• Nicolai Maass 1 ,
• Philipp Elischer 3 ,
• Ann Carolin Longardt 3 ,
• Karolin Tesch 4 ,
• Annett Lebenatus 4 ,
• Magret Krüger 5 &
• Ibrahim Alkatout 1  

Journal of Medical Case Reports volume  18 , Article number:  196 ( 2024 ) Cite this article

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Introduction

The management of a pregnancy in a bicornuate uterus is particularly challenging. A bicornuate uterus is a rare occurrence and a twin pregnancy in a bicornuate uterus even more rare. These pregnancies call for intensive diagnostic investigation and interdisciplinary care.

Case presentation

We report on a 27-year-old European woman patient (gravida I, para 0) with a simultaneous pregnancy in each cavity of a bicornuate bicollis uterus after embryo transfer. The condition was confirmed by hysteroscopy and laparoscopy. Several unsuccessful in vitro fertilization (IVF) attempts had been performed earlier before embryo transfer in each cornus. After a physiological course of pregnancy with differential screening at 12 + 6 weeks and 22 + 0 weeks of gestation, the patient presented with therapy-resistant contractions at 27 + 2 weeks. This culminated in the uncomplicated spontaneous delivery of the leading fetus and delayed spontaneous delivery of the second fetus.

Only 16 cases of twin pregnancy in a bicornuate unicollis uterus have been reported worldwide and only 6 in a bicornuate bicollis uterus. The principal risks in such pregnancies are preterm labor, intrauterine growth restriction, malpresentation and preeclampsia. These typical risk factors of a twin pregnancy are greatly potentiated in the above mentioned setting.

A twin pregnancy in the presence of a uterine malformation is rare and difficult to manage. These rare cases must be collected and reported in order to work out algorithms of monitoring and therapy as well as issue appropriate recommendations for their management.

Peer Review reports

Pregnancy and delivery in women with Müllerian anomalies are associated with numerous complications, including preterm labor and the need for greater attention to achieve successful parturition. The prevalence of congenital uterine anomalies varies among different populations: it is 5.5% in the general population, 8.0% in women with infertility, and 13.3% in women with a history of abortions. It peaks at 24.5% among patients with a history of abortions as well as infertility [ 1 ].

The presence of a uterine anomaly is known to be associated with adverse pregnancy outcomes, including a higher risk of spontaneous abortion, preterm labor, cesarean delivery due to breech presentation, and reduced live births when compared to a normal uterus [ 2 ]. Nevertheless, the frequency of these outcomes varies across different types of uterine anomalies. Several classifications have been proposed, such as the vagina cervix uterus adnex-associated malformation (VCUAM) classification [ 3 ], the classification of the American Society of Reproductive Medicine (ASRM) [ 4 ] or the European Society of Human Reproduction and Embryology (ESHRE) and the European Society of Gynaecological Endoscopy (ESGE) [ 5 ]. A classification by the ASRM was established in a revision of the American Fertility Society (AFS) classification from 1988 (Fig.  1 ) [ 6 ].

Printable version of the Müllerian Anomalies Classification Table by Pfeifer et al . The Müllerian anomaly described in this review is marked with a star [ 6 ]

The current prevalence of congenital uterine anomalies is a mere 5.5–6.7% in the general population, 7.3% in sterile women, and approximately 16.7% in women with recurrent miscarriage [ 1 , 7 ]. The majority of bicornuate uteruses are asymptomatic and typically detected during a routine evaluation of the patient. In cases of more severe symptoms, such as primary amenorrhea, hematocolpos, pelvic pain, dyspareunia or obstacle during vaginal penetration, the condition is diagnosed early [ 7 ]. Otherwise, a uterine malformation is first diagnosed during obstetric complications, including spontaneous abortions, intrauterine growth restriction, preterm rupture of membranes, preterm labor, or malpresentation of the infant [ 8 ].

A bicornuate uterus is a rare congenital uterine malformation. A mere 16 cases of twin pregnancy associated with a bicornuate unicollis uterus have been reported worldwide, and most of these were delivered with a cesarean section [ 9 , 10 ].

Pregnancies in the bicornuate uterus are typically regarded as a high-risk condition due to their association with adverse reproductive outcomes, including recurrent pregnancy loss, cervical insufficiency, low birthweight, preterm birth, malpresentation, cesarean delivery, and uterine rupture [ 2 ].

To our knowledge, this is the first report of a simultaneous pregnancy in each cavity of a bicornuate bicollis uterus and vaginal delivery of twins in the 28th week of pregnancy. The malformation was known prior to IVF and the embryo transfer was deliberately performed into one uterine horn to increase the likelihood of pregnancy.

We report a 27-year-old European woman patient, gravida 1, para 1 at 27 + 2 weeks of gestation, with dichorionic diamniotic twin pregnancy and a bicornuate bicollis uterus. In 2021 the patient had undergone diagnostic laparoscopy with chromopertubation and hysteroscopy because she wished to have children. The procedure revealed a bicornuate bicollis uterus with two separate cervical canals arising from one portio. The uterus was seen on laparoscopy with a heart-shaped external contour (Fig.  1 , marked with a star), V0 C1 U2 A0 M0 per the VCAUM classification (Fig.  2 ). After six unsuccessful inseminations and one frustrated intracytoplasmic sperm injection, parallel embryo transfer into one uterine horn each was performed in February 2023. This resulted in successful implantation of the embryos in one uterine horn each. The subsequent course of pregnancy history was initially physiological. The patient had an unremarkable first-trimester and second-trimester screening.

Vagina Cervix Uterus Adnex-associated Malformation classification by the European Society of Human Reproduction and Embryology/European Society of Gynaecological Endoscopy from Grimbizis et al . [ 5 ]

On 4 August 2023 the patient developed contractions and cervical shortening to 10 mm in the leading pregnancy. She received the first antenatal steroid prophylaxis consisting of 12 mg celestane under nifedipine tocolysis with supersaturation, and was then transferred to our level III perinatal center. On admission she continued to be symptomatic, with a prolapsed amniotic sac in the leading pregnancy and an otherwise unremarkable concordant dichorionic diamniotic twin pregnancy (first fetus: vertex presentation, estimated weight 916 g, left lateral placenta; second fetus: vertex presentation, estimated weight 1161 g left lateral placenta) with physiological amniotic fluid and fetal/fetomaternal Doppler findings. The tocolytic agent was switched to atosiban. The patient received antibiotic therapy with cefuroxime 1.5 g intravenously according to the regimen and a single oral dose of 1 g azithromycin for prolapse of the amniotic sac. She was admitted to the delivery room for further cardiotocography (CTG) monitoring. Detailed discussions were held with the patient about the possible mode of delivery. In view of all potential risks (including emergency cesarean section, cesarean section on the second fetus after spontaneous delivery of the first, atony, hysterectomy), we decided on a spontaneous delivery of the leading fetus with a possible wait-and-watch approach in regard of the second. The patient was informed verbally and in writing about the potential need for an emergency cesarean section and hysterectomy with bilateral salpingectomy. Our neonatologists and anesthesiologists also informed the patient about the subsequent procedures and the risks of preterm birth. Red blood cell concentrates were kept on call. With increasing labor activity, intravenous magnesium as neuroprotection was started per schedule. Nine hours later the patient presented with strong contractions, a sensation of pressure, and prolapse of the amniotic sac to the middle of the vagina. Tocolysis with atosiban was stopped, an amniotomy performed, and a male infant was born with the short-term support of an oxytocin infusion. With a birthweight of 1150 g (P 69), an Apgar score of 9/9/9, an arterial umbilical cord pH of 7.29, and base excess -7.70 mmol/l, the infant was transferred immediately to the neonatologists. The placenta followed spontaneously, the anesthesiology and surgery nursing team were sent away, and the patient remained well in the delivery room under further CTG monitoring. The ultrasound investigation revealed an empty right cavum while the left cervix appeared long and closed. In order to avoid uterine atony, the recommencement of tocolysis was initially postponed; the patient was currently labor free. After three hours her unstoppable contractions started again. With a pushing contraction she delivered the second infant: male, 1115 g (P 62), Apgar score 8/7/9, an arterial umbilical cord pH of 7.36, and base excess − 7.80 mmol/l. The infant was immediately transferred to the neonatologists. Three units of oxytocin as a short infusion were given and the placenta followed spontaneously. On ultrasound both uterine horns were empty and there was no birth injury or hemorrhage. The mother could be discharged after three days in good health.

Postnatally the first-born twin suffered from respiratory distress syndrome and was therefore given a surfactant twice by less invasive surfactant application (LISA) within the first 24 h after birth. The infant’s respiratory situation remained stable thereafter under non-invasive airway support. A patent ductus arteriosus (PDA) was treated successfully with ibuprofen. The clinical surveillance remained uneventful in regard of enteral feeding and there was no intraventricular bleeding.

The second twin also needed surfactant treatment for respiratory distress syndrome. A routine chest X-ray on the first day of the infant’s life suggested a sacculation of the esophagus, demanding endotracheal intubation for airway protection. Detailed imaging after transesophageal administration of a contrast agent revealed a remarkably large esophageal sacculation. The examination showed no fistulation towards the surrounding tissue or organs and no other signs of malformation. The newborn was extubated and a stable respiratory situation was maintained by non-invasive respiratory support. Next, endoscopic esophageal examination was performed to establish the need for any therapeutic intervention. A hemodynamically significant PDA was treated with ibuprofen and paracetamol. To date, a minimal residual PDA is being monitored closely and has revealed no hemodynamic irregularities. Similar to the twin brother, intracranial examinations in the second infant were uneventful and enteral feeding was established without complications.

Figures  3 and 4 are intraoperative images of the uterine malformation on laparoscopy and an ultrasound performed during parturition.

Intraoperative recording of hysteroscopy and laparoscopy, a intraoperative view of two separate cervical canals arising from one portio. b intraoperative view of a heart-shaped uterus and physiological adnexa

Ultrasound examination for control of contraction of the right uterine horn as well as exclusion of placental remnants after delivery of the first pregnancy

The patient underwent a checkup, a 3D ultrasound investigation, and an MRI of the pelvis at eight weeks after delivery to confirm the diagnosis of a uterine malformation established previously by other colleagues. After a regular puerperium, the patient was in a good general condition. The speculum examination revealed an inconspicuous singular portio and the bimanual examination showed an enlarged anteflexed uterus with otherwise unremarkable findings. Three-dimensional ultrasound revealed a bicornuate uterus with a prominent septum extending to the portio (Fig.  5 a, b). The ovaries as well as the kidneys appeared physiological on 2D ultrasound.

3D ultrasound at 8 weeks postpartum; a Bicornuate bicollis uterus (functional unicollis) in the transverse plane with both uterine cavities. b Bicornuate bicollis uterus (functional unicollis): cross-section through both uterine cavities, 3D ultrasound

A magnetic resonance imaging (MRI) of the pelvis confirmed our diagnosis of a functional bicornuate unicollis uterus with the postpartum condition of a residual, partly strong cervical septum apically, which possibly ruptured peripartum (Fig.  6 a–d). Therefore, our diagnosis according to the VCUAM classification was V0 C + U2 A0 M0, a bicornuate uterus with a functional unicollis and a prominent septum.

Magnetic resonance imaging at 8 weeks postpartum; a Bicornuate bicollis uterus (functional unicollis) in the transverse plane with both uterine cavities (transverse T2-weighted sequence, Avanto 1.5 T, Siemens Healthineers). b Bicornuate bicollis uterus (functional unicollis): cross-section through both uterine cavities near the fundus (coronary T2-weighted sequence, Avanto 1.5 T Siemens Healthineers). c Bicornuate bicollis uterus (functional unicollis): cross-section through both uterine cavities near the isthmus (coronary T2-weighted sequence, Avanto 1.5 T Siemens Healthineers). d Bicornuate bicollis uterus (functional unicollis): cross-section through the uterine cervix with the postpartum condition of a residual, partly strong cervical septum apically—possibly ruptured peripartum (coronary T2-weighted sequence, Avanto 1.5 T Siemens Healthineers)

The case described here is a rarity in terms of fertility medicine as well as obstetrics, with an exemplary successful outcome. Correct diagnosis of a uterine anomaly in the context of infertility, accurate conclusions, and IVF treatment after previously unsuccessful fertility attempts are important steps. Basic knowledge of uterine anomalies is a prerequisite for making correct decisions in each case. The next step is intensified pregnancy monitoring and knowledge of the existing risks. The final step is planning birth with due consideration to the risks involved.

A thorough understanding of embryology is essential when dealing with congenital anomalies of the uterus. The uterus, cervix, fallopian tubes and upper third of the vagina develop from the fused Müllerian ducts or paramesonephric ducts. Initially, fusion of the ducts is incomplete and a septum is present within the lumina. However, as time progresses the septum disappears and a single large cavity is formed [ 11 , 12 ]. A bicornuate unicollis uterus is a congenital uterine malformation typically arising from incomplete fusion of the Müllerian ducts, leading to a varying degree of separation between the uterine cavities [ 12 , 13 ]. Incomplete fusion of the Müllerian or paramesonephric ducts results in a bicornuate bicollis uterus, characterized by a double or single vagina, double cervices, and two single‐horned uteruses with partial fusion of their muscular walls. Renal anomalies as well as a vaginal septum may occur in these cases. In contrast, a uterus didelphys is seen in cases of complete lack of fusion of the Müllerian ducts, characterized in the majority of cases by two uterine cavities and two cervices with a longitudinal vaginal septum [ 12 , 14 ].

Advanced investigation techniques such as MRI and 2D ultrasonography have made it easier to diagnose a malformed uterus [ 15 ]. Transvaginal 3D ultrasonography proved to be highly accurate in diagnosing and classifying congenital uterine anomalies, surpassing both hysteroscopy and MRI in its effectiveness. However, the management of uterine malformations is not a fixed procedure and varies according to the individual's clinical history [ 16 , 17 ].

Uterine malformations may significantly impact pregnancy outcomes. Therefore, the misdiagnosis of such malformations must be avoided.

Women with a malformation of the uterus are confronted with poor chances of a normal pregnancy because their uterus does not offer the suitable physiological environment of a normal pear-shaped uterus. This condition may lead to obstetric complications such as infertility, ectopic pregnancy, and prematurity. Consequently, women with congenital uterine malformations have a low probability of conceiving naturally, causing the clinician and the patient to consider IVF. Surprisingly, after IVF/ Intracytoplasmic sperm injection (ICSI) treatment Kong et al . reported no difference in pregnancy rates between a bicornuate uterus and a normal uterus [ 18 ]. Chan et al . also reported that bicornuate uteruses do not reduce fertility. Women with a bicornuate uterus have a moderately increased risk of miscarriage, preterm birth and fetal malpresentation [ 19 ].

Currently, there is no established approach for the management of this type of pregnancy. Although vaginal delivery is not contraindicated, a cesarean section was performed in all 16 reported cases [ 20 ]. The reason for a cesarean section was an urgent indication for delivery, such as preeclampsia [ 8 , 9 ] or the avoidance of obstetric complications such as malpresentation and uterine rupture [ 13 , 21 , 22 ]. A cesarean section was performed by two separate uterotomies in the lower uterine segment [ 9 , 21 , 22 ] or a bilateral vertical incision [ 14 ].

A preventive cerclage of the cervix may be considered, but is known to be associated with a poor reproductive outcome in uterine malformations [ 8 ]. Likewise, the National Institute for Health and Care Excellence (NICE) guidelines do not recommend a routine cervical cerclage to prevent spontaneous preterm birth in women with a twin or triplet pregnancy [ 23 , 24 ].

Chemlal et al . reported a case of spontaneous twin pregnancy in a bicornuate unicollis uterus. The authors performed a preventive cervical cerclage with non-absorbable sutures at 12 weeks, and final delivery by cesarean section due to preeclampsia at 35 weeks [ 8 ].

Karunaratne et al . described a case of twin pregnancy in a patient with a bicornuate bicollis unterus and breech presentation of the fetus, delivered by primary cesarean section at 37 + 0 weeks. They also reviewed the literature and discussed several relevant points such as the feasibility of vaginal delivery versus cesarean section, the surgical approach in cesarean section, and delivery timing. They found that a cesarean section is preferentially used in cases of malpresentation, fetal distress and labor dystocia. The authors also give preference to longitudinal uterotomies. On the other hand, bilateral vertical incisions are recommended in small and externally fused lower uterine segments in order to avoid extension, injury to the septum, difficult repair and hemorrhage, and facilitate future pregnancies [ 14 ]. In the assumed presence of two functional uteruses and independent induction of labor, it would be advisable to adopt a two-stage approach. After the first delivery one would wait in order to prolong the second pregnancy and avoid a preterm birth. This approach has been successfully used in bicornuate bicollis uterus [ 25 ] as well as uterus didelphys [ 26 ].

The optimal follow-up for twin pregnancies in patients with Müllerian fusion anomalies remains a subject of controversy, given the risks and the rarity of these cases. The treatment must be tailored to the individual case. Figure  7 summarizes our checklist for comprehensive and intensive monitoring of patients with twin pregnancy in a bicornuate uterus.

Comprehensive and intensive monitoring of patients with twin pregnancy in a bicornuate uterus

In our case, the postpartum 3D ultrasound and MRI led us to conclude that the correct classification of the uterine malformation was a bicornuate bicollis uterus, functional unicollis with a prominent septum extending to the portio. Our classification of the malformation—a functional unicollis rather than bicollis—would have led us to attempt spontaneous delivery. However, a wait-and-watch approach with interval delivery would not have been considered promising in view of the facts. Our current knowledge of the uterine anomaly offered the patient the option of performing a septum dissection later in order to raise the chances of spontaneous conception as well as reduce the risk of abortion and preterm birth.

Uterine malformations must be detected early in order to prevent obstetric complications. Ultrasound, especially 3D ultrasound, and MRI play an important role in the detection of malformations. Twin pregnancy in a bicornuate uterus is very rare and bears a high risk of obstetric complication such as fetal growth restriction or preterm birth. Intensive monitoring is essential for successful management of these rare pregnancies. According to the guidelines, twin pregnancies should be monitored by serial ultrasound every two weeks in order to assess fetal growth and measure cervical length, and thus determine the risk of intrauterine growth restriction and premature delivery. However, we lack guidelines for monitoring these pregnancies and the mode of delivery.

Despite the rarity of this condition, a primary caesarean section should not be selected by default. The patient should be informed about the different delivery options and their risks. The mode of delivery should be determined individually. If spontaneous parturition is the goal, the clinician and the patient must be prepared for possible complications including emergency caesarean section and atony, and take interdisciplinary precautions that will avoid unnecessary delay in emergency situations.

These rare cases must be collected and reported in order to work out algorithms of monitoring and therapy, and provide suitable recommendations to the patients for an optimum outcome of pregnancy.

Data availability

The datasets analyzed for the current study are available from the corresponding author upon reasonable request.

Abbreviations

American Fertility Society

American Society of Reproductive Medicine

Body Mass Index

Cardiotocography

European Society of Gynaecological Endoscopy

European Society of Human Reproduction and Embryology

In vitro fertilization

Less invasive surfactant application

Magnetic resonance imaging

National Institute for Health and Care Excellence

Patent ductus arteriosus

Vagina Cervix Uterus Adnex-associated Malformation

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Acknowledgements

We acknowledge financial support from the province of Schleswig-Holstein as a part of the Open Access Publication Fond (DFG-OA-Fonds) funding program.

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Department of Obstetrics and Gynecology, University Hospital of Schleswig Holstein, Campus Kiel, 24105, Kiel, Germany

Melanie Schubert, Kristin Andresen, Zino Ruchay, André Farrokh, Nicolai Maass & Ibrahim Alkatout

Department of Obstetrics and Gynecology, University Hospital of Würzburg, 97080, Würzburg, Germany

Anastasia Tihon

Department for Pediatrics and Adolescent Medicine I, Neonatology, University Hospital of Schleswig Holstein, Campus Kiel, 24105, Kiel, Germany

Philipp Elischer & Ann Carolin Longardt

Department of Radiology and Neuroradiology, University Hospital Schleswig-Holstein, Campus Kiel, 24105, Kiel, Germany

Karolin Tesch & Annett Lebenatus

Center for Operative Gynecology, Park Clinic, 24116, Kiel, Germany

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Conceptualization, M.S. and I.A.; Project administration, M.S., A.F. and I.A.; Supervision, M.S. and I.A.; Visualization, I.A., A.T., K.A., Z.R., A.F. and N.M.; Writing—original draft preparation, M.S., A.T., I.A., A.C.L., P.E.; Writing—review and editing, M.S. and I.A. All authors have read and agreed to the published version of the manuscript.

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Schubert, M., Tihon, A., Andresen, K. et al. Peripartal management of dichorial twin pregnancy in a bicornuate bicollis uterus: a case report and review of the literature. J Med Case Reports 18 , 196 (2024). https://doi.org/10.1186/s13256-024-04506-2

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Received : 09 January 2024

Accepted : 12 March 2024

Published : 21 April 2024

DOI : https://doi.org/10.1186/s13256-024-04506-2

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• Dichorial twin pregnancy
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• Bicornuate bicollis uterus
• Uterine malformations
• Preterm birth

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