Reproductive Failure in Males with Cystic Fibrosis

Kaplan, Elvin; Shwachman, Harry; Perlmutter, Alan D.; Rule, Allyn H.; Khaw, Kon‐Taik; Holsclaw, Douglas S.

doi:10.1056/nejm196807112790203

Cited by 344 publications

(146 citation statements)

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“…In the literature, the proportion of men with CBAVD and impaired spermatogenesis ranges from 12 to 35% (Kaplan et al, 1968;Oates & Amos, 1994;Okada et al, 1999;Viville et al, 2000;Weiske et al, 2000;Meng et al, 2001). The literature definitions of spermatogenesis in studies of CBAVD have variously been based on clinical experience (Meng et al, 2001), Johnsen's score (Okada et al, 1999), Valette's score (Valette et al, 1976;Viville et al, 2000) and De Kretser and Holstein's classification (De Kretser & Holstein, 1976;Weiske et al, 2000).…”

Section: Discussionmentioning

confidence: 99%

“…Otherwise, many studies have evaluated the quality of the spermatogenesis in CBAVD obstruction although it is generally assumed that sperm production is normal in CBAVD. Thus, histopathological studies of the spermatogenesis quality from CBAVD patients have generated divergent results, with either normal or altered spermatogenesis (Kaplan et al, 1968;Goldstein & Schlossberg, 1988;Silber et al, 1990;Gottlieb et al, 1991;Okada et al, 1999;Viville et al, 2000;Weiske et al, 2000;Meng et al, 2001).However, it is not known whether the outcomes of ICSI depend on the quality of spermatogenesis in men with CBAVD.…”

Section: Introductionmentioning

confidence: 99%

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Congenital bilateral absence of the vas deferens: the impact of spermatogenesis quality on intracytoplasmic sperm injection outcomes in 108 men

et al. 2015

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SUMMARYIn azoospermic men with congenital bilateral absence of the vas deferens (CBAVD), it is not known whether the outcomes of intracytoplasmic sperm injection (ICSI) depend on the quality of testicular spermatogenesis (as determined histopathologically). We retrospectively studied the impact of spermatogenesis quality on ICSI outcomes in 108 azoospermic men with CBAVD consulting in a university hospital's department of andrology and reproductive biology. As part of an ICSI program, sperm samples were obtained from the epididymis [by microsurgical epididymal sperm aspiration (MESA); n = 47] or the testis [by testicular sperm extraction (TESE); n = 14] or both (MESA + TESE, n = 47). In the TESE group (i.e., TESE-only and MESA + TESE), spermatogenesis was normal in 21 of the 108 men (19.4%) and hypospermatogenesis occurred in 33 (30.5%). The fertilization rate was significantly lower in the hypospermatogenic group than in the normospermatogenesis group (65.6 and 72.9%, respectively; p = 0.02); this was also true for the embryo cleavage rate (88.6 and 92.1%, respectively; p = 0.007), and the proportion of embryos with fewer than 30% of enucleate fragments (79.5 and 86.9%, respectively; p = 0.02). Our study results showed that impaired spermatogenesis had a negative impact on certain early-stage biological outcomes of ICSI. In CBAVD, male factors are likely to exert a harmful effect on the early stages of embryo development.

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Section: Discussionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

Congenital bilateral absence of the vas deferens: the impact of spermatogenesis quality on intracytoplasmic sperm injection outcomes in 108 men

et al. 2015

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“…L'exploration 6chographique permet de mettre en 6vidence une anomalie des v6si-cutes s6minales plus d'l fois sur 2. Enfin~ l'6chographie r6nale met en 6vidence une ag6-n6sie r6nale homolat6rale ~ une ag6n6sie de la v6sicule s6minale dans 20 % des cas [6][7][8][9][10].…”

Section: Abcd Et Clinique [14]unclassified

“…Dans la s6rie publi6e par DUMUR [4], 50 % des tests de la sueur sont positifs (>60mEq/1), ce qui laisse pr6sager que probablement 50 % sont des h6t6rozygotes composites. En revanche on ne retrouve jamais de mutation chez les patients avec ag6n6sie r6na-le (20 %) [4][5][6][7][8][9][10]. S'agit-il d'un processus pathologique diff6rent ?…”

Section: Genetique Et Abcdunclassified

L’agenesie bilaterale congenitale des deferents a l’heure de I’ICSI

Rigot

Gervais

Dumur³

et al. 1998

Androl.

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RESUME L'ag4n4sie bilatdrale congdnitale des d4f4rents est ranomalie la plus fr4quente des voies g4nitales masculines. Le diagnostic est clinique, le bilan spermiologique associe azoospermie, hypospermie, pH acide, marqueurs 6pididymaires bas. Le bilan morphologique recherche une anomalie des v4sicules s4minales et une ag4n4sie r4nale. Une mutation du g~ne CFTR est retrouv4e dans plus de 70 % des eas. Un conseil g6n@tique s'impose avant toute aide m4dicale ~ la procr4a-tion. Les r@sultats de rICSI avec sperme prdlev4 chirurgicalement sont les m~mes que dans les autres causes d'azoospermies excr4toires. Mots clefs : azoosperrnie, agdndsie des ddfdrents, mucoviscidose, ICSIL'ag6n6sie bilat6rale cong6nitale des d6f6-rents (ABCD) est la malformation la plus fr6-quente des voies s4minales. Sa fr6quence est 6valu6e ~ 0,5 % dans les s6ries autopsiques, 1% chez des patients infertiles, 6valu6s entre 17 et 30 % chez les patients pr6sentant une azoospermie excr6toire [14]. Elle repr6sente une situation de st6rilit6 masculine a priori d6finitive. En 1952, YOUNC devait proposer les premiers spermatoc61es et quelques cas de grossesses devaient @tre d6crits. En 1985, TEMPLE, SMITH ouvraient d'autres perspectives en r6alisant une f6condation in vitro avec sperme pr61ev6 chirurgicalement. Enfin, I'ICSI permettait d'envisager des succ6s jusque-l~ inesp6r6s et une prise en charge de l'infertilit4 des hommes mucovicidosiques. En effet, ces hommes 6talent connus comme porteurs d'une ag6n6sie cong6nitale des d6f6rents quasi constante. Se posait ainsi directement le lien entre l'ag6n4sie bilat6rale cong~nitale des d6f6rents et la mucoviscidose. Cette hypoth6se avait d6j~ 6t6 6voqu6e par LE LANNOU [9]. DUMUR [3] en montrant la fr4quence anormalement 61ev6e de la mutation h F508 chez des patients st6riles avec une ABCD, validait cette hypoth6se qui 6tait confirm6e de fa~on d6finitive par ANGUIANO [1] en 1992 qui mettait en 6vidence des h6t6rozygotes composites chez des patients st6riles avec une ABCD. L'ABCD devenait ainsi le premier exemple de st6rilit6 masculine d6finitive d'origine g6n6-tique curable par les techniques d'aide m@di-cale ~ la procr6ation avec un risque de transmission d'une maladie potentiellement grave au conceptus. EMBRYOLOGIE DES VOIES SEMINALES NORMALES [2]Les voies g6nitales sont issues des canaux m6son6phrotiques de Wolf et param6son6-phrotiques de Mullerl Ces structures sont elles-m@mes sous la d6pendance du facteur antimullerien et la testost6rone testicutaire foetale et de son d6riv6 la dihydrotestost6rone. Ainsi les canaux param6son6phrotiques dis-57

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“…It is known that cystic fibrosis and primary ciliary dyskinesia (immotile cilia syndrome) are the most commoncauses for the combination of male infertility and sinopulmonary infections in Japan (1)(2)(3)(4). However, although Young's syndrome is not so rare in Caucasians, there have been only a few reports of this syndrome in Asia, and it is extremely rare among Japanese (5)(6)(7).…”

Section: Introductionmentioning

confidence: 99%

A Rare Case of Young's Syndrome in Japan

Hasegawa

Ohe²,

Yamazaki³

et al. 1994

Intern. Med.

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A 56-year-old man was admitted for the evaluation of recurrent pulmonary infections. Since the patient presented with the combination of diffuse cystic bronchiectasis, chronic sinusitis and obstructive azoospermia, the diagnosis ofYoung's syndromewas made. Because the sweat chloride value, pancreatic function and ciliary ultrastructure were normal, this case could be clearly differentiated from cystic fibrosis and primary ciliary dyskinesia. There have been only a few reported cases of Young's syndrome in Japan. (Internal Medicine 33: 649-653, 1994)

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Reproductive Failure in Males with Cystic Fibrosis

Cited by 344 publications

References 4 publications

Congenital bilateral absence of the vas deferens: the impact of spermatogenesis quality on intracytoplasmic sperm injection outcomes in 108 men

Congenital bilateral absence of the vas deferens: the impact of spermatogenesis quality on intracytoplasmic sperm injection outcomes in 108 men

L’agenesie bilaterale congenitale des deferents a l’heure de I’ICSI

A Rare Case of Young's Syndrome in Japan

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