Jejunal cavernous lymphangioma manifested as gastrointestinal bleeding with hypogammaglobulinemia in adult: A case report and literature review

Table 1 Summary of small bowel lymphangioma cases in adults from 1955 to 2019

Ref.	Age/ gender	Ethnicity/ country	Symptoms	Laboratory results	Imaging/ endoscopy	Location	Layer/ appea-rance	Number/ size	Treatment	Pathology
Present case	29/F	Asian/ China	Melena, fatigue, palpitations after activities	Anemia: Hgb 6.8 g/dL; IgG 2.91 g/L	Neg: EGD, CLS, CTE, PET/CT Pos: 99mTC-RBC-S, DBE	Mid-jej	SM/LP, elevated lesion with white lymphatic dilatation changes and fresh blood stains	1/3 cm × 2 cm + 1/ satellite	Surgery	Cavernous Lpgm
Samuelson et al[4], 2018	70/M	Caucasian/American	Weight loss, hemate-mesis, abdominal pain,	—	Pos: CT (intussus-ception)	Jej	—	1/2.5 cm	Surgery	Lpgm
Lim et al[1], 2018	70/M	Asian/ South Korea	Melena, dizziness	Anemia: Hgb 5.2 g/dL	Pos: ES	Prox-jej	SM; yellow-white polyploids with bleeding	Mu/small	Surgery (TT)	Lpgm
Huang et al[5], 2017	58/M	Asian/ Taiwanese	Melena/ dyspnea/ dizziness	Anemia: Hgb 3.5 g/dL	Neg: EGD, CLS; Pos: CT, SBE	3rd Duo	Soft, yellow-pink, lobulated mass	1/15 cm × 15 cm	Surgery (Lap)	Lpgm
Ng et al[6], 2016	33/M	Asian/ Singapore	Episodic melena, dyspnea, lethargy	Anemia: Hgb 3.9 g/dL	Neg: EGD, CLS; Pos: CT, SBE	Prox-jej	LP/SM; “strawberry” polyploids	1/2.2 cm × 2.0 cm	Surgery (Lap, TT)	Cavernous Lpgm
Tseng et al[7], 2016	88/F	Asian/ China Taiwan	Episodic melena, fatigue, shortness of breath	Anemia: Hgb 8 g/dL	Neg: EGD, CLS; Pos: CE, SBE	Dis-Duo, Prox-jej	Lobular circumfe-rential	1/2.8 cm × 2.4 cm+Mu/satellite	Surgery (Lap)	Cavernous Lpgm
Limaiem et al[8], 2015	22/F	Caucasian/Tunis	intermittent colicky abdominal pain, diarrhea	Anemia	Pos: CT/US (intussus-ception)	Jej	SM; peduncu-lated polyp	1/2 cm	Surgery (Lap)	Lpgm
Bucciero et al[9], 2015	28/M	Caucasian/ Italy	Melena	Anemia: Hgb 4.0 g/dL	Pos: CT (inhomo-geneous mass)	3rd Duo	Circumferential ulcerated with bleeding	Mu/small	Surgery	Lpgm
Antonino et al[10], 2014	24/F	Caucasian/ Italy	Melena	Anemia: Hgb 6.8 g/dL	Pos: EGD, MRI	2nd Duo	Ulcerated polypoid	1/5 cm	Surgery	Hemo-Lpgm
Kida et al[11], 2012	81/F	Asian/ Japan	Melena	Anemia:Hgb 5.0 g/dl	Neg: EGD, CLS, CT; Pos: CE	Prox-jej	SM/LP; white-yellowish peduncu-lated polyp	1/2 cm × 1 cm	Polypec-tomy+hemoclipping+APC (DBE)	Lpgm
Fang et al[12], 2012	57/F	Asian/ China	Melena	Anemia	Neg: EGD; Pos: ES	Prox-jej	—	1/5 cm × 4 cm	Surgery	Hemo-Lpgm
Mavrogenis et al[13], 2011	26/F	Caucasian/ France	Melena	Anemia	Neg: EGD, CLS, CT; Pos: CE	Prox-jej	Whitish carpet-like villi and superficial red spots	1/ circumference 2/3	Surgery (Lap)	Hemo-Lpgm
Mavrogenis et al[13], 2011	59/F	Caucasian/ France	Melena	Anemia	Neg: EGD, CLS; Pos: CE	Prox-jej	Polypoid covered whitish and red spots	1/3.5 cm × 7 cm	Surgery	Hemo-Lpgm
Morris-Stiff et al[14], 2011	34/M	Caucasian/American	Dyspnea	Anemia: Hgb 6 g/dL	Pos: CT, CE, ES	Prox-jej	SM; lobular circumfe-rential	1/5.3 cm × 4 cm × 1.5 cm	Surgery (TT)	Lpgm
Huang et al[15], 2009	57/M	Caucasian/American	Melena, shortness of breath	Anemia: Hgb 7.6 g/dL	Neg: EGD, CLS; Pos:CE, DBE	Mid-jej	Firm mass with central ulceration	1/3.5 cm × 2.2 cm	Surgery (Lap)	Carvernous Lpgm
Hsu et al[16], 2007	75/M	Asian/ China Taiwan	Melena	Anemia	Neg: EGD, CLS, BR, CT; Pos: 99mTC-RBC-S, DBE	Dis-jej	SM; circumfe-rential	1/6 cm × 4 cm	Surgery (Lap)	Lpgm
Griffa et al[17], 2006	54/M	Caucasian/ Italy	Episode of melaena	Anemia	Neg: EGD, CLS, US; Pos: CE	Mid-jej	SM; white-yellowish polypoid	1/1.5 cm × 2 cm	Surgery (Lap)	Lpgm
Honda et al[18], 2003	31/F	Asian/ Japan	Melena	Anemia: Hgb 5.3 g/dL	Neg: EGD, CLS; Pos: BR, CT, ES	Prox-jej	Lobulated, yellowish-white surface with red and white specks	1/3.5 cm × 3 cm	Surgery	Lpgm
Ishikawa et al[19], 1999	64/M	Asian/Japan	Melena	Anemia	Pos: BR,DBE	Upper-Ile	SM	1/5.5 cm × 3 cm	Surgery	Cavernous Lpgm
Barquist et al[20], 1997	33/F	Caucasian/Panama	Chronic gastrointe-stinal blood	Anemia: Hgb 5.5 g/dL	Neg: EGD; Pos: ES	Mid-Jej	SM	1/3.3 cm × 2.5 cm × 1.0 cm	Surgery (Lap)	Cavernous Lpgm
Hanagiri et al[21], 1992	53/M	Asian/ Japan	Bowel obstruction	Hgb 11 g/dL	Pos: CT	Jej	SM, smooth and yellowish	1/2.5 cm × 2 cm	Surgery (Lap)	Cavernous Lpgm
Colizza et al[22], 1981	58/F	Caucasian/ Italy	Melena	Anemia: Hgb 7.5 g/dL	Neg: BR	SMH, Prox-jej	SM/LP	2/4 cm × 5 cm	Surgery (Lap)	Cystic Lpgm
Ralston[23], 1961	58/M	Caucasian/Australian	Gastrointe-stinal bleeding	Anemia: Hgb 6.6 g/dL	Neg: BR,EGD	Prox-jej	SM	1/1 cm × 0.5 cm	Surgery	Hemo-Lpgm

99mTC-RBC-S: 99m Technetium labeled red blood cells scintigraphy; APC: argon plasma coagulation; BR: barium radiography; CE: Capsule endoscopy; CLS: colonoscopy; CT: Computed tomography; CTE: Computed tomography enterography; DBE: Double-balloon enteroscopy; Dis-jej: distal jejunum; Duo: duodenal; EGD: Esophagogastroduodenoscopy; ES: Enteroscopy; Hemo-Lpgm: hemolymphangiomas; Hgb: hemoglobin; Ile: ileum; LP: lamina propria; Lap: laparoscopic; Lpgm: lymphangiomas; Mid-jej: middle jejunum; Mu: multiple; Neg: negative; PET/CT: Positron-emission tomography/computed tomography; Pos: positive; Prox-jej: proximal jejunum; SB: small bowel; SBE: single-balloon enteroscopy; SM: submucosa; SMH: stomach; TT: tattooing marked; US: ultrasonography.