Recurrent chronic histiocytic intervillositis with intrauterine growth restriction, osteopenia, and fractures

Crawford, Anthony C.; Moore, Lynette; Bennett, Gregg; Savarirayan, Ravi; Manton, Nicholas; Khong, Yee; Barnett, Christopher; Haan, Eric

doi:10.1002/ajmg.a.37856

Cited by 16 publications

(6 citation statements)

References 18 publications

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“…12 A report also suggests that CHI may mimic features of osteogenesis imperfecta, including bone fractures in the foetus, though this was unable to be confirmed genetically. 13 Three pregnancies studied in this report resulted in FGR and a small placenta, suggesting that foetal development may have been limited by uteroplacental insufficiency. CHI reportedly affects 6 in 10 000 s and third trimester placentas sent for histopathological examination 14 and has been identified in 4.4% of first trimester miscarriages with normal karyotype.…”

mentioning

confidence: 75%

Chronic histiocytic intervillositis: A breakdown in immune tolerance comparable to allograft rejection?

Brady

Williams

Sharps

et al. 2020

American J Rep Immunol

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From implantation of the embryo through to birth, successful pregnancy relies upon the promotion of a tolerogenic uterine environment to allow the foetus to thrive. 1 Simultaneously, and of equal importance, the maternal inflammatory response must be conserved to protect the mother and foetus against pathogens. 2 This "paradox" of pregnancy, first described by Sir Peter Medawar in the 1950s, 1 has prompted investigation into the immunology of pregnancy, particularly where it may be dysfunctional. Chronic histiocytic intervillositis (CHI), also known as chronic intervillositis (CI), chronic intervillositis of unknown aetiology

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mentioning

confidence: 75%

Chronic histiocytic intervillositis: A breakdown in immune tolerance comparable to allograft rejection?

Brady

Williams

Sharps

et al. 2020

American J Rep Immunol

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“…When assessing birth trauma, it is important to consider underlying fetal conditions that can mimic inflicted trauma, such as connective tissue disorders in lacerations (44), bone disease in fractures (45 –47), hematological conditions in bruises and intracranial hemorrhages (48), and the role of asphyxia in the latter (3, 26, 49). Extensive subaponeurotic hemorrhage may cause death due to hypovolemic shock (42, 50).…”

Section: Discussionmentioning

confidence: 99%

Forensic Aspects of Perinatal Deaths

Cohen¹,

Scheimberg²

2018

Academic Forensic Pathology

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From a forensic pathologist's perspective, there are several aspects of the perinatal postmortem that are particularly important. If a fetus is found abandoned, the pathologist needs to ascertain the fetal age, the appropriateness of growth, if the baby was born alive or dead, and the possible causes of death. In cases of litigation for perinatal deaths occurring in hospitals, access to the obstetric and neonatal notes (if the baby is born alive and dies a few hours or days later) is fundamental to reach a correct interpretation and conclusion. The most important points to consider in cases of intrapartum death are the roles of asphyxia and trauma in the causation of the baby's death. Timing of the fetal death in relation to delivery may also be an important point in these cases. Finally, intrapartum lesions should always be considered in the differential diagnosis of possible child abuse in babies aged two months or less.

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“…However, other evidence implies that the primary problem is maternal, particularly in the case of CHI. Firstly, CHI carries a recurrence rate of up to 100% and worsens in successive pregnancies (36,(81)(82)(83). Secondly, when women with recurrent pregnancy loss due to CHI undergo IVF with their own oocyte and their partner's sperm, followed by implantation into a surrogate, CHI does not recur (84).…”

Section: Chronic Placental Inflammation and Autoimmune Diseasementioning

confidence: 99%

Chronic Inflammatory Placental Disorders Associated With Recurrent Adverse Pregnancy Outcome

2022

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Chronic inflammatory placental disorders are a group of rare but devastating gestational syndromes associated with adverse pregnancy outcome. This review focuses on three related conditions: villitis of unknown etiology (VUE), chronic histiocytic intervillositis (CHI) and massive perivillous fibrin deposition (MPFD). The hallmark of these disorders is infiltration of the placental architecture by maternal immune cells and disruption of the intervillous space, where gas exchange between the mother and fetus occurs. Currently, they can only be detected through histopathological examination of the placenta after a pregnancy has ended. All three are associated with a significant risk of recurrence in subsequent pregnancies. Villitis of unknown etiology is characterised by a destructive infiltrate of maternal CD8+ T lymphocytes invading into the chorionic villi, combined with activation of fetal villous macrophages. The diagnosis can only be made when an infectious aetiology has been excluded. VUE becomes more common as pregnancy progresses and is frequently seen with normal pregnancy outcome. However, severe early-onset villitis is usually associated with fetal growth restriction and recurrent pregnancy loss. Chronic histiocytic intervillositis is characterised by excessive accumulation of maternal CD68+ histiocytes in the intervillous space. It is associated with a wide spectrum of adverse pregnancy outcomes including high rates of first-trimester miscarriage, severe fetal growth restriction and late intrauterine fetal death. Intervillous histiocytes can also accumulate due to infection, including SARS-CoV-2, although this infection-induced intervillositis does not appear to recur. As with VUE, the diagnosis of CHI requires exclusion of an infectious cause. Women with recurrent CHI and their families are predisposed to autoimmune diseases, suggesting CHI may have an alloimmune pathology. This observation has driven attempts to prevent CHI with a wide range of maternal immunosuppression. Massive perivillous fibrin deposition is diagnosed when >25% of the intervillous space is occupied by fibrin, and is associated with fetal growth restriction and late intrauterine fetal death. Although not an inflammatory disorder per se, MPFD is frequently seen in association with both VUE and CHI. This review summarises current understanding of the prevalence, diagnostic features, clinical consequences, immune pathology and potential prophylaxis against recurrence in these three chronic inflammatory placental syndromes.

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Recurrent chronic histiocytic intervillositis with intrauterine growth restriction, osteopenia, and fractures

Cited by 16 publications

References 18 publications

Chronic histiocytic intervillositis: A breakdown in immune tolerance comparable to allograft rejection?

Chronic histiocytic intervillositis: A breakdown in immune tolerance comparable to allograft rejection?

Forensic Aspects of Perinatal Deaths

Chronic Inflammatory Placental Disorders Associated With Recurrent Adverse Pregnancy Outcome

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