Abdominal kaposiform hemangioendothelioma presenting as volvulus and recurring intestinal obstruction in a neonate: a case report

Abstract Background Kaposiform hemangioendothelioma is a rare vascular tumor primarily occurring in infants and children. The most common sites for kaposiform hemangioendothelioma are extremities, with very few cases of abdominal kaposiform hemangioendothelioma reported in neonates. Making a diagnos...

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Main Authors: Salaar Ahmed, Bakhtawar Dilawar, Sarosh Moeen, Shahzadi Dilawar, Hafsa Sheikh, Muhammad Anwar Arain
Format: Article
Language:English
Published: BMC 2024-12-01
Series:Journal of Medical Case Reports
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Online Access:https://doi.org/10.1186/s13256-024-04976-4
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author Salaar Ahmed
Bakhtawar Dilawar
Sarosh Moeen
Shahzadi Dilawar
Hafsa Sheikh
Muhammad Anwar Arain
author_facet Salaar Ahmed
Bakhtawar Dilawar
Sarosh Moeen
Shahzadi Dilawar
Hafsa Sheikh
Muhammad Anwar Arain
author_sort Salaar Ahmed
collection DOAJ
description Abstract Background Kaposiform hemangioendothelioma is a rare vascular tumor primarily occurring in infants and children. The most common sites for kaposiform hemangioendothelioma are extremities, with very few cases of abdominal kaposiform hemangioendothelioma reported in neonates. Making a diagnosis of Kaposiform hemangioendothelioma can be challenging when the patient presents with generalized symptoms such as bilious vomiting and constipation that can be attributed to other more common causes of intestinal obstruction. Case presentation A 28-day-old Pakistani baby girl presented with bilious vomiting and constipation. The abdomen was distended, and scans showed generalized dilation of bowel loops. A preliminary diagnosis of intestinal obstruction due to volvulus was made, and the patient underwent an exploratory laparotomy. Postoperatively, the patient remained unwell with profuse aspirates in nasogastric and no improvement in ileus. Repeat scans were again suggestive of midgut volvulus, which was initially managed conservatively, but later redo surgery was done. Ischemic bowel was resected with histopathology showing kaposiform hemangioendothelioma. In the meantime, the baby developed an abdominal collection, infection, and short gut syndrome due to resection of the bowel. The family was counseled regarding the prognosis, and the patient was transferred from hospital to home upon request. The baby passed away 2 days later. Conclusion While extremely rare, abdominal kaposiform hemangioendothelioma can be an important and potentially lifesaving differential diagnosis to consider in neonates and infants with recurring and nonresolving intestinal obstruction. The management is complex, and prognosis can be poor in diffuse, congested lesions involving a large part of the gut.
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spelling doaj-art-606efa55b89742fbb89f462437203d222025-08-20T01:59:44ZengBMCJournal of Medical Case Reports1752-19472024-12-011811610.1186/s13256-024-04976-4Abdominal kaposiform hemangioendothelioma presenting as volvulus and recurring intestinal obstruction in a neonate: a case reportSalaar Ahmed0Bakhtawar Dilawar1Sarosh Moeen2Shahzadi Dilawar3Hafsa Sheikh4Muhammad Anwar Arain5Medical College, Aga Khan UniversityDepartment of Surgery, Aga Khan UniversityDepartment of Pathology, Aga Khan UniversityNational Institute of Child HealthDepartment of Surgery, Aga Khan UniversityDepartment of Surgery, Aga Khan UniversityAbstract Background Kaposiform hemangioendothelioma is a rare vascular tumor primarily occurring in infants and children. The most common sites for kaposiform hemangioendothelioma are extremities, with very few cases of abdominal kaposiform hemangioendothelioma reported in neonates. Making a diagnosis of Kaposiform hemangioendothelioma can be challenging when the patient presents with generalized symptoms such as bilious vomiting and constipation that can be attributed to other more common causes of intestinal obstruction. Case presentation A 28-day-old Pakistani baby girl presented with bilious vomiting and constipation. The abdomen was distended, and scans showed generalized dilation of bowel loops. A preliminary diagnosis of intestinal obstruction due to volvulus was made, and the patient underwent an exploratory laparotomy. Postoperatively, the patient remained unwell with profuse aspirates in nasogastric and no improvement in ileus. Repeat scans were again suggestive of midgut volvulus, which was initially managed conservatively, but later redo surgery was done. Ischemic bowel was resected with histopathology showing kaposiform hemangioendothelioma. In the meantime, the baby developed an abdominal collection, infection, and short gut syndrome due to resection of the bowel. The family was counseled regarding the prognosis, and the patient was transferred from hospital to home upon request. The baby passed away 2 days later. Conclusion While extremely rare, abdominal kaposiform hemangioendothelioma can be an important and potentially lifesaving differential diagnosis to consider in neonates and infants with recurring and nonresolving intestinal obstruction. The management is complex, and prognosis can be poor in diffuse, congested lesions involving a large part of the gut.https://doi.org/10.1186/s13256-024-04976-4Kaposiform hemangioendotheliomaBowel obstructionVolvulusNeonatePediatric surgery
spellingShingle Salaar Ahmed
Bakhtawar Dilawar
Sarosh Moeen
Shahzadi Dilawar
Hafsa Sheikh
Muhammad Anwar Arain
Abdominal kaposiform hemangioendothelioma presenting as volvulus and recurring intestinal obstruction in a neonate: a case report
Journal of Medical Case Reports
Kaposiform hemangioendothelioma
Bowel obstruction
Volvulus
Neonate
Pediatric surgery
title Abdominal kaposiform hemangioendothelioma presenting as volvulus and recurring intestinal obstruction in a neonate: a case report
title_full Abdominal kaposiform hemangioendothelioma presenting as volvulus and recurring intestinal obstruction in a neonate: a case report
title_fullStr Abdominal kaposiform hemangioendothelioma presenting as volvulus and recurring intestinal obstruction in a neonate: a case report
title_full_unstemmed Abdominal kaposiform hemangioendothelioma presenting as volvulus and recurring intestinal obstruction in a neonate: a case report
title_short Abdominal kaposiform hemangioendothelioma presenting as volvulus and recurring intestinal obstruction in a neonate: a case report
title_sort abdominal kaposiform hemangioendothelioma presenting as volvulus and recurring intestinal obstruction in a neonate a case report
topic Kaposiform hemangioendothelioma
Bowel obstruction
Volvulus
Neonate
Pediatric surgery
url https://doi.org/10.1186/s13256-024-04976-4
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