Fetal hypoxia secondary to severe maternal anemia as a causative link between blueberry muffin baby and erythroblastosis: a case report

Abstract

BackgroundNeonatal blueberry muffin lesions are rare cutaneous eruptions, presenting as transient, non-blanching, red-violaceous papules, mostly localized in the trunk, head and neck, attributable to a marked dermal hematopoietic activity. Congenital infections of the TORCH complex (toxoplasmosis, other, rubella, cytomegalovirus and herpes) and hematological disorders have been classically associated with this neonatal dermatological manifestation. We report for the first time an unusual presentation of blueberry muffin lesions in a neonate born from a mother affected by severe anemia during pregnancy.Case presentationA male, white Caucasian, neonate showed a cutaneous rash at birth, suggestive of “blueberry muffin”-like lesions. These cutaneous lesions were associated with marked elevation of the circulating nucleated red blood cells, and with ultrasound findings of peculiar brain ischemic porencephalic lesions. The clinical features of spontaneous disappearance and the association with marked erythroblastosis strongly suggest that these dermatological findings may be the consequence of an extramedullary hematopoiesis unexpectedly evoked by the intrauterine chronic exposure to hypoxia caused by severe maternal anemia.ConclusionsIn conclusion, fetal hypoxia secondary to severe maternal anemia may play a causative and unreported role in the development of neonatal blueberry muffin lesions.

Blueberry muffin syndrome in a newborn

“…Дифференциальная диагностика кожных высыпаний у новорожденных является сложной задачей, поскольку причиной кожной сыпи у новорожденного могут быть самые разнообразные состояния, такие как врожденные инфекции, перинатальная анемия, вызванная гемолитической болезнью (несовместимость по Rh-фактору, системе ABO), наследственный сфероцитоз, фето-фетальный трансфузионный синдром, фетоплацентарная недостаточность, внутричерепное кровоизлияние, нейродегенеративные заболевания (синдром Айкарди-Гутьерес), неонатальная красная волчанка, новообразования и аутоиммунные или гематологические нарушения [8,9,14,15].…”

Extramedullary hematopoiesis secondary to COVID-19: A first case report in a newborn

“…[1][2][3] Most frequent are infections of the TORCH complex (toxoplasmosis, rubeola, cytomegalovirus, herpes). 4,5 This case was unique in that the mother had a confirmed case of COVID-19 (positive PCR test) shortly before giving birth, and this was the only risk factor explaining why COVID-19 was the likely cause of extramedullary hematopoiesis.…”

Cutaneous extramedullary haematopoiesis: Implications in human disease and treatment

“…[46] A recent case identified blueberry muffin lesions in a patient with foetal erythroblastosis and hypoxia secondary to severe maternal anaemia. [47] Other blueberry muffin rashes associated with anaemia include CEMH in the setting of Rh haemolytic disease of the newborn [48] and hereditary spherocytosis. [39,49] Blueberry muffin rashes have also been associated with congenital disorders such as Aicardi-Goutieres syndrome [50] and congenital non-ketotic hyperglycinemia.…”

“…[58] Another patient presented with truncal papules followed by facial subcutaneous nodules. [59] [44] Idiopathic myelofibrosis [52,53] Haemolytic disorder [39,49] Polycythemia vera [67] ABO incompatibility [45] Essential thrombocytopenia [68] Rh incompatibility [48] Primary hypertrophic osteoarthropathy [69] Twin-twin transfusion [45] Chronic myelogenous leukaemia [73] Maternal anaemia [47] Pilomatricoma [70] Inborn errors of metabolism [41,45] diagnosed as tri-lineage CEMH have been reported. [60,61] One patient who underwent splenectomy developed bullae at the surgical scar that spread centrifugally within days to cover the abdomen, groin, chest and back.…”

“…One additional patient demonstrated ABO incompatibility and another resulted from twin‐twin transfusion syndrome, a cause previously reported in the literature . A recent case identified blueberry muffin lesions in a patient with foetal erythroblastosis and hypoxia secondary to severe maternal anaemia . Other blueberry muffin rashes associated with anaemia include CEMH in the setting of Rh haemolytic disease of the newborn and hereditary spherocytosis .…”