Subarachnoid hemorrhage due to nonbranching aneurysm of the middle cerebral artery in a young adult with a history of Kawasaki disease

Abstract

Background:The incidence of subarachnoid hemorrhage (SAH) in young adults is relatively rare. Kawasaki disease is a systemic vasculopathy that is known to cause coronary artery aneurysms; however, its effect on cerebral arteries remains largely unclear.Case Description:We report the case of a 20-year-old male with a history of Kawasaki disease who presented with SAH caused by the rupture of a nonbranching middle cerebral artery aneurysm. This is the third report of SAH associated with Kawasaki disease. Preoperative echocardiography of the patient rejected the presence of bacterial endocarditis and other heart abnormalities. An emergency craniotomy and clip occlusion of the aneurysm was successfully performed without obstructing the parent artery. Two weeks later, the patient was discharged without any apparent neurological deficit. We also performed a circumstantial pathological study on specimens obtained from the aneurysm wall. Our histological findings suggest that the elastic lamina and tunica intima were completely destroyed during the acute vasculitis phase of Kawasaki disease, which possibly led to the aneurysmal formation.Conclusions:Lack of active inflammatory changes and atherosclerotic lesions may explain the chronic feature of Kawasaki disease, not a typical aneurysmal formation.

Neurological Involvement in Primary Systemic Vasculitis

“…However, although rare cases of first onset have also been reported in adult patients, most reported cases are in young adults and the manifestations are due to late-onset sequelae instead of new-onset active disease. We found one case (47) describing a 20-year-old young adult with a history of KD suffering from subarachnoid hemorrhage due to an intracranial aneurysm with a stalk-like narrow neck, located at the trunk of the middle cerebral artery. As the researchers mentioned, coronary aneurysms in KD also arise in places where no branch exists.…”

A Case of Complete Adult-Onset Kawasaki Disease: A Review of Pathogenesis and Classification

“…Classical KD is unusual in adults, with 81 cases previously reported in the literature since 2010 and 13 cases of incomplete adult-onset KD [1,3,9,10]. Our patient fulfilled the criteria for the classical (typical) KD.…”

Involvement of Nervous System in Kawasaki Disease

“…In general, most authors agree that neurological complications in Kawasaki disease reflect an increased underlying inflammatory burden. 10,13 15 Ishida et al, 26 Sabatier et al, 16 Yamazaki-Nakashimada et al, 25 Wang et al, 17 Ahn et al, 24 Tanaka et al, 28 Wada et al, 22 Du et al, 27 Suda et al, 18 Hoshino et al, 29 Fujiwara et al, 19 Laxer et al, 20 51 Stowe, 50 Khubchandani and Dhanrajani, 49 Kocabas et al, 48 Thapa, 47 Kaur et al, 46 Lim et al, 45 Li et al, 43 Wright et al, 44 Larralde et al, 42 Poon et al 52 Abducens nerve palsy 4 (case reports) Emiroglu et al, 54 Rodriguez-Lozano et al, 55 Guven et al, 56 Wurzburger et al 57 Articles identified by our literature search consisted mostly of case reports or case series. Prospective and retrospective studies are uncommon and can be attributed to the relative rarity of neurological complications in this entity.…”

“…Clinical symptoms usually include headache, altered mental status, gait disturbances, as well as pyramidal signs during neurological examination. [24][25][26][27][28][29] It is noteworthy that in three of the above cases (a 4-year-old boy, an adolescent, and a young adult), stroke did not occur during acute phase but at 2, 9, and 18 years, respectively, after Kawasaki diagnosis. All these patients had received intravenous immunoglobulins.…”

“…All these patients had received intravenous immunoglobulins. 25,26,28 The under-lying mechanism in the above cases included rupture of cerebral vessel aneurysms. It should also be noted that aneurysm formation in cerebral vessels of these patients may be revealed in later stages.…”