Lupus anticoagulant hypoprothrombinemia syndrome associated with bilateral adrenal haemorrhage in a child: early diagnosis and intervention View Full Text


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Article Info

DATE

2021-03-17

AUTHORS

Atsushi Sakamoto, Masao Ogura, Atsushi Hattori, Kinji Tada, Reiko Horikawa, Hisaya Nakadate, Kimikazu Matsumoto, Keiji Nogami, Masahiro Ieko, Akira Ishiguro

ABSTRACT

BackgroundLupus anticoagulant-hypoprothrombinemia syndrome (LAHPS) is characterized by bleeding and thrombosis in patients with autoimmune diseases or infections. Paediatric LAHPS exhibits various degrees of bleeding, ranging from mild to severe; however, adrenal haemorrhage due to LAHPS and its long-term clinical course have not been sufficiently described.Case presentationA 9-year-old boy presented with prolonged abdominal pain and abnormal coagulation screening tests. The laboratory tests showed prolonged activated partial thromboplastin time and subsequently revealed the presence of lupus anticoagulant, anti-nuclear antibodies, and hypoprothrombinemia, leading to diagnosis of LAHPS. An enhanced computed tomogram demonstrated nodular lesions in the adrenal glands bilaterally, suggestive of adrenal haemorrhage. Laboratory and clinical manifestations exhibited life-threatening adrenal insufficiency that required hydrocortisone administration. The patient developed systemic lupus erythematosus, diagnosed 12 months later.ConclusionsThis patient with LAHPS developed rare adrenal failure due to adrenal haemorrhage, a life-threatening event that should be recognized and treated early. In our case, renal dysfunction was also observed when systemic lupus erythematosus was diagnosed 1 year after LAHPS. Our case emphasizes that early recognition of adrenal failure and careful long-term observation is required in patients with autoantibodies. More... »

PAGES

19

Identifiers

URI

http://scigraph.springernature.com/pub.10.1186/s12959-021-00271-0

DOI

http://dx.doi.org/10.1186/s12959-021-00271-0

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https://app.dimensions.ai/details/publication/pub.1136468130

PUBMED

https://www.ncbi.nlm.nih.gov/pubmed/33731125


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