Medical and Health Sciences
Medical and Health Sciences
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Pregnancy after Intracytoplasmic Sperm Injection in Couples with Kartagener Syndrome: Two Clinical Cases.

Gravidez pós-ICSI em casais com Síndrome de Kartagener: A propósito de dois casos clínicos.

Miranda, C.*, Costa, S.*, Coelho, D.* & Dantas, S.*
*) Hospital Senhora da Oliveira, Guimarães, Portugal.
Filmed: 
2021-09-25
Submitted: 
2022-08-29
Published: 
2022-09-14

CHAPTERS

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CITATION

Miranda, C., Costa, S., Coelho, D., & Dantas, S. (2021, September 24-25). Pregnancy after intracytoplasmic sperm injection in couples with Kartagener syndrome: two clinical cases [Paper presentation]. XXXVII Reproductive Studies Journeys, Lisbon, Portugal. https://academy-on.com/videos/mh-373-11539
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SYNOPSIS

This communication presents two cases of Kartagener syndrome where pregnancy was achieved with ICSI. Spermatozoa selection was performed using different methods and high fertilization rates were achieved in both cases. Our work highlights the importance of evaluation of spermatozoa by an experienced embryologist as one of the crucial steps in the management of these couples.
Esta comunicação apresenta dois casos de Síndrome de Kartagener em que a gravidez foi obtida com ICSI. A seleção de espermatozoides foi realizada usando diferentes técnicas, tendo-se obtido elevadas taxas de fertilização em ambos os casos. O nosso trabalho realça a importância da avaliação dos espermatozoides por um embriologista experiente como um dos passos fundamentais na abordagem destes casais.
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ABSTRACT

Primary ciliary dyskinesia (PCD) is an inherited disease characterized by anomalies in the ciliary structure or function causing congenital impairment of mucociliary clearance. Kartagener syndrome (KS) is a rare subtype of PCD defined by the triad: bronchiectasis, chronic sinusitis and situs inversus. KS has an estimated prevalence of 1 in 20.000-40.000 and inheritance is usually autosomal recessive. Male patients with KS have almost no spontaneous pregnancies due to complete immotility of their spermatozoa. Intracytoplasmic sperm injection (ICSI) is considered the treatment of choice despite showing lower fertilization rates in these patients. Different tests for assortment and selection of viable immotile spermatozoa are described. We present two cases of Kartagener syndrome where pregnancy was achieved with ICSI using different methods of spermatozoa selection.
A Discinesia Ciliar Primária (DCP) é uma doença hereditária caracterizada por anomalias na estrutura ou função dos cílios levando a alterações na clearance mucociliar. A Síndrome de Kartagener (SK) é um subtipo raro de DCP definida pela tríade: bronquiectasias, sinusite crónica e situs inversus. A SK tem uma prevalência estimada de 1 em 20.000-40.000 e na maioria das vezes apresenta um padrão hereditário autossómico recessivo. Homens com SK apresentam uma probabilidade de gravidez espontânea quase nula devido à imobilidade dos espermatozoides. A injeção intracitoplasmática de espermatozoides (ICSI) é considerada o tratamento de escolha nestes casais, apesar de apresentar taxas de fertilização menor nestes pacientes. Diferentes testes para avaliação e seleção de espermatozoides imóveis viáveis são descritos. Apresentamos dois casos de SK em que a gravidez foi conseguida com ICSI usando diferentes métodos de seleção de espermatozoides.
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KEYWORDS

Reproductive Techniques, Assisted; Kartagener Syndrome; Sperm Injections, Intracytoplasmic; Ciliary Motility Disorders; Asthenozoospermia.
Técnicas de reprodução assistida; Síndrome Kartagener; Injeção Intracitoplasmática de Espermatozóides; Anomalia motilidade ciliar; Astenozoospermia.
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DISCLOSURES

The author certifies that there is no actual or potential conflict of interest in relation to this publication.
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REFERENCES

Abu-Musa A, Hannoun A, Khabbaz A, Devroey P. (1999) Failure of fertilization after intracytoplasmic sperm injection in a patient with Kartagener’s syndrome and totally immotile spermatozoa: case report. Hum Reprod.14(10):2517-8. doi:10.1093/humrep/14.10.2517 Kaushal M, Baxi A. (2007) Birth after intracytoplasmic sperm injection with use of testicular sperm from men with Kartagener or immotile cilia syndrome. Fertil Steril. 88(2):497.e9-11. doi:10.1016/j.fertnstert.2006.11.113 Abu-Musa A, Nassar A, Usta I. (2008) In vitro fertilization in two patients with Kartagener’s syndrome and infertility. Gynecol Obstet Invest. 65(1):29-31. doi: 10.1159/000106503 Yildirim G, Ficicioglu C, Akcin O, Attar R, Tecellioglu N, Yencilek F. (2009) Can pentoxifylline improve the sperm motion and ICSI success in the primary ciliary dyskinesia? Arch Gynecol. 279(2):213-5. doi: 10.1007/s00404-008-0671-y Matsumoto Y, Goto S, Hashimoto H, Kokeguchi S, Shiotani M, Okada H. (2010) A healthy birth after intracytoplasmic sperm injection using ejaculated spermatozoa from a patient with Kartagener’s syndrome. Fertil Steril. 93(6):2074.e17-9. doi: 10.1016/j.fertnstert.2009.11.042 Sha YW, Ding L, Li P. (2014) Management of primary ciliary dyskinesia/Kartagener’s syndrome in infertile male patients and current progress in defining the underlying genetic mechanism. Asian J Androl. 16(1):101-6. Doi: 10.4103/1008-682X.122192 Tadesse A, Alemu H, Silamsaw M, Gebrewold Y. (2018) Kartagener’s syndrome: a case report. J Med Case Rep. 12(1):5. 10.1186/s13256-017-1538-2 Ozkavukcu S, Celik-Ozenci C, Konuk E, Atabekoglu C. (2018) Live birth after Laser Assisted Viability Assessment (LAVA) to detect pentoxifylline resistant ejaculated immotile spermatozoa during ICSI in a couple with male Kartagener’s syndrome. Reprod Biol Endocrinol. 16(1):10. doi:10.1186/s12958-018-0321-6. doi: 10.1186/s12958-018-0321-6 Aamir J, Ashwini LS, Ganguly D, Murugan S, Muthiah SS, Kainoth S, Reddy GR. (2015) Interpretation: Real Time Assessment on Immotile but Viable Spermatozoa for Intracytoplasmic Sperm Injection (ICSI): an Embryologists Outlook. Austin J In Vitro Fertili. 2(3): 1021. Nordhoff V. (2015) How to select immotile but viable spermatozoa on the day of intracytoplasmic sperm injection? An embryologist’s view. Andrology. 3(2): 156-62. doi: 10.1111/andr.286
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AUTHOR BIOGRAPHY

Cláudia Miranda graduated in Medicine by Instituto Ciência Biomédicas Abel Salazar do Porto in 2015. Worked as a general residente in Centro Hospitalar Póvoa de Varzim/ Vila do Conde during 2016. Started her specialization in Gynecology and Obstetrics in 2017 at Hospital Senhora da Oliveira, Guimarães, where she is currently working. Has a particular interest in endoscopic surgery, fertility and vulvovaginal disease.

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