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. 2024 May 3;10(1):110.
doi: 10.1186/s40792-024-01884-z.

Laparoscopic excision of accessory spleen for recurrent autoimmune hemolytic anemia after splenectomy: a case report

Ryosuke Kashiwagi  1   2 , Masaharu Ishida  1 , Koichi Onodera  3 , Shuichi Aoki  1 , Masahiro Iseki  1 , Takayuki Miura  1 , Hideo Ohtsuka  1 , Masamichi Mizuma  1 , Kei Nakagawa  1 , Takashi Kamei  1 , Michiaki Unno  4
Affiliations

Laparoscopic excision of accessory spleen for recurrent autoimmune hemolytic anemia after splenectomy: a case report

Ryosuke Kashiwagi et al. Surg Case Rep. .

Abstract

Background: Splenectomy is indicated in cases of autoimmune hemolytic anemia (AIHA), which are refractory to medical management. In post-splenectomy, there exists a theoretical risk of AIHA recurrence, especially if an accessory spleen undergoes compensatory hypertrophy. In this context, we present a unique case of recurrent AIHA managed through laparoscopic excision of the accessory spleen (LEAS).

Case presentation: A 60-year-old male underwent laparoscopic splenectomy (LS) for AIHA refractory to standard medical therapies. Following the surgery, there was a marked improvement in hemolytic anemia symptoms, and oral steroid therapy was terminated 7 months post-LS. Nonetheless, a year after the LS, the patient exhibited a marked decline in hemoglobin levels, dropping to a concerning 5.8 g/dl, necessitating the reintroduction of oral steroids. A subsequent contrast-enhanced computed tomography (CT) scan unveiled an enlarged accessory spleen. The patient then underwent LEAS, during which the accessory spleen, obscured within adipose tissue, proved challenging to visualize laparoscopically. This obstacle was surmounted utilizing intraoperative ultrasonography (US), enabling successful excision of the accessory spleen. The post-surgical period progressed without complications, and the steroid dosage was reduced to one-twelfth of its initial preoperative quantity.

Conclusions: Recurrent AIHA can be instigated by post-splenectomy compensatory hypertrophy of the accessory spleen. Ensuring comprehensive splenic tissue excision is crucial in AIHA management to obviate recurrent stemming from hypertrophic remnants. In scenarios of AIHA recurrence tied to an enlarged accessory spleen, LEAS stands as a viable and effective therapeutic modality.

Keywords: AIHA; Autoimmune hemolytic anemia; Intraoperative ultrasonography; Laparoscopic excision of accessory spleen.

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Conflict of interest statement

The authors declare no competing interests.

Figures

Fig. 1
Fig. 1
HYPERLINK "sps:id::fig1||locator::gr1||MediaObject::0"Clinical progression timeline: following the initial LS, steroid dosage was tapered from 15 mg/day to 5 mg/day. Anemia re-emerged a year post-surgery, prompting the LEAS procedure. After the second surgery, steroid dosage was tapered from 60 mg/day to 5 mg/day. Hb hemoglobin. Ret reticulocyte. T-Bil total-bilirubin. HP haptoglobin. LS laparoscopic splenectomy. LEAS laparoscopic excision of accessory spleens. PSL prednisolone, 60 mg is equivalent to 1 mg/kg for this patient
Fig. 2
Fig. 2
Contrast-enhanced CT scan depicting accessory spleen hypertrophy: prior to the initial LS, the accessory spleen measured 7 mm (arrow). It expanded to 16 mm upon AIHA recurrence (arrowhead). A pre-laparoscopic splenectomy; B at AIHA recurrence
Fig. 3
Fig. 3
Accessory spleen imaging via SPIO-enhanced MRI: the MRI highlighted a marked signal reduction (arrow) on the SPIO-enhanced T2 and T2* weighted images (WI). A T2WI, B SPIO-enhanced T2WI, C T2*WI, D: SPIO-enhanced T2*WI
Fig. 4
Fig. 4
Surgical photograph and intraoperative ultrasonography illustrating the accessory spleen: during the procedure, ultrasonography identified the accessory spleen. Accessory spleen, arrow; left kidney, arrowhead
See this image and copyright information in PMC

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