Infantile hepatic hemangioma and hepatic mesenchymal hamartoma in an infant associated with placental mesenchymal dysplasia: a case report
Abstract Background Although infantile hepatic hemangioma and hepatic mesenchymal hamartoma are relatively common in benign pediatric liver tumors, coexistence of the two tumors is rare. Placental mesenchymal dysplasia is also a rare disorder. We report the case of a baby girl born after a pregnancy...
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Japan Surgical Society
2022-08-01
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| Series: | Surgical Case Reports |
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| Online Access: | https://doi.org/10.1186/s40792-022-01519-1 |
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| author | Shunsuke Fujii Kyoko Mochizuki Hidehito Usui Norihiko Kitagawa Sayoko Umemoto Mio Tanaka Yukichi Tanaka Masako Otani Kumiko Nozawa Kenji Kurosawa Masayo Kagami Masato Shinkai |
| author_facet | Shunsuke Fujii Kyoko Mochizuki Hidehito Usui Norihiko Kitagawa Sayoko Umemoto Mio Tanaka Yukichi Tanaka Masako Otani Kumiko Nozawa Kenji Kurosawa Masayo Kagami Masato Shinkai |
| author_sort | Shunsuke Fujii |
| collection | DOAJ |
| description | Abstract Background Although infantile hepatic hemangioma and hepatic mesenchymal hamartoma are relatively common in benign pediatric liver tumors, coexistence of the two tumors is rare. Placental mesenchymal dysplasia is also a rare disorder. We report the case of a baby girl born after a pregnancy complicated by placental mesenchymal dysplasia, who developed both infantile hepatic hemangioma and hepatic mesenchymal hamartoma. Case presentation The patient was born at 32 weeks and 5 days of gestation for impending placental abruption, weighing 1450 g. Liver tumors, composed of both hypervascular solid and large cystic lesions, were detected after birth and markedly increased to create abdominal distention within 9 months. Diagnostic imaging suspected the coexistence of infantile hepatic hemangioma and cystic hepatic mesenchymal hamartoma. Following propranolol therapy for infantile hepatic hemangioma and needle puncture of a large cyst, the cystic lesions and adjacent hypervascular lesions were partially resected via laparotomy. Pathological findings confirmed the coexistence of hepatic mesenchymal hamartoma and infantile hepatic hemangioma, which had no association with androgenetic/biparental mosaicism. The postoperative course was uneventful, and the tumor had not regrown after 3 years. Conclusions Although the coexistence of infantile hepatic hemangioma and hepatic mesenchymal hamartoma associated with placental mesenchymal dysplasia is extremely rare, the pathological and pathogenetic similarities between these disorders suggest that they could have derived from similar embryologic origins rather than being a mere coincidence. Further follow-up is required, with careful attention to the potential for malignant hepatic mesenchymal hamartoma transformation. |
| format | Article |
| id | doaj-art-63cda1b417954b28b81c7c5cd57a97b8 |
| institution | DOAJ |
| issn | 2198-7793 |
| language | English |
| publishDate | 2022-08-01 |
| publisher | Japan Surgical Society |
| record_format | Article |
| series | Surgical Case Reports |
| spelling | doaj-art-63cda1b417954b28b81c7c5cd57a97b82025-08-20T02:51:52ZengJapan Surgical SocietySurgical Case Reports2198-77932022-08-01811610.1186/s40792-022-01519-1Infantile hepatic hemangioma and hepatic mesenchymal hamartoma in an infant associated with placental mesenchymal dysplasia: a case reportShunsuke Fujii0Kyoko Mochizuki1Hidehito Usui2Norihiko Kitagawa3Sayoko Umemoto4Mio Tanaka5Yukichi Tanaka6Masako Otani7Kumiko Nozawa8Kenji Kurosawa9Masayo Kagami10Masato Shinkai11Department of Surgery, Kanagawa Children’s Medical CenterDepartment of Surgery, Kanagawa Children’s Medical CenterDepartment of Surgery, Kanagawa Children’s Medical CenterDepartment of Surgery, Kanagawa Children’s Medical CenterDepartment of Pathology, Kanagawa Children’s Medical CenterDepartment of Pathology, Kanagawa Children’s Medical CenterDepartment of Pathology, Kanagawa Children’s Medical CenterDepartment of Diagnostic Pathology, Yokohama City University Medical CenterDepartment of Radiology, Kanagawa Children’s Medical CenterDepartment of Medical Genetics, Kanagawa Children’s Medical CenterDepartment of Molecular Endocrinology, National Research Institute for Child Health and DevelopmentDepartment of Surgery, Kanagawa Children’s Medical CenterAbstract Background Although infantile hepatic hemangioma and hepatic mesenchymal hamartoma are relatively common in benign pediatric liver tumors, coexistence of the two tumors is rare. Placental mesenchymal dysplasia is also a rare disorder. We report the case of a baby girl born after a pregnancy complicated by placental mesenchymal dysplasia, who developed both infantile hepatic hemangioma and hepatic mesenchymal hamartoma. Case presentation The patient was born at 32 weeks and 5 days of gestation for impending placental abruption, weighing 1450 g. Liver tumors, composed of both hypervascular solid and large cystic lesions, were detected after birth and markedly increased to create abdominal distention within 9 months. Diagnostic imaging suspected the coexistence of infantile hepatic hemangioma and cystic hepatic mesenchymal hamartoma. Following propranolol therapy for infantile hepatic hemangioma and needle puncture of a large cyst, the cystic lesions and adjacent hypervascular lesions were partially resected via laparotomy. Pathological findings confirmed the coexistence of hepatic mesenchymal hamartoma and infantile hepatic hemangioma, which had no association with androgenetic/biparental mosaicism. The postoperative course was uneventful, and the tumor had not regrown after 3 years. Conclusions Although the coexistence of infantile hepatic hemangioma and hepatic mesenchymal hamartoma associated with placental mesenchymal dysplasia is extremely rare, the pathological and pathogenetic similarities between these disorders suggest that they could have derived from similar embryologic origins rather than being a mere coincidence. Further follow-up is required, with careful attention to the potential for malignant hepatic mesenchymal hamartoma transformation.https://doi.org/10.1186/s40792-022-01519-1Hepatic infantile hemangiomaHepatic mesenchymal hamartomaPlacental mesenchymal dysplasiaPropranololTumorectomy |
| spellingShingle | Shunsuke Fujii Kyoko Mochizuki Hidehito Usui Norihiko Kitagawa Sayoko Umemoto Mio Tanaka Yukichi Tanaka Masako Otani Kumiko Nozawa Kenji Kurosawa Masayo Kagami Masato Shinkai Infantile hepatic hemangioma and hepatic mesenchymal hamartoma in an infant associated with placental mesenchymal dysplasia: a case report Surgical Case Reports Hepatic infantile hemangioma Hepatic mesenchymal hamartoma Placental mesenchymal dysplasia Propranolol Tumorectomy |
| title | Infantile hepatic hemangioma and hepatic mesenchymal hamartoma in an infant associated with placental mesenchymal dysplasia: a case report |
| title_full | Infantile hepatic hemangioma and hepatic mesenchymal hamartoma in an infant associated with placental mesenchymal dysplasia: a case report |
| title_fullStr | Infantile hepatic hemangioma and hepatic mesenchymal hamartoma in an infant associated with placental mesenchymal dysplasia: a case report |
| title_full_unstemmed | Infantile hepatic hemangioma and hepatic mesenchymal hamartoma in an infant associated with placental mesenchymal dysplasia: a case report |
| title_short | Infantile hepatic hemangioma and hepatic mesenchymal hamartoma in an infant associated with placental mesenchymal dysplasia: a case report |
| title_sort | infantile hepatic hemangioma and hepatic mesenchymal hamartoma in an infant associated with placental mesenchymal dysplasia a case report |
| topic | Hepatic infantile hemangioma Hepatic mesenchymal hamartoma Placental mesenchymal dysplasia Propranolol Tumorectomy |
| url | https://doi.org/10.1186/s40792-022-01519-1 |
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