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Table 1 Review of literature on isolated hypoganglionosis from 1978 to 2009

From: Isolated adult hypoganglionosis presenting as sigmoid volvulus: a case report

First author/reference No. of cases Specimen Staining Treatment Mortality
Zhang et al. [5] 17 Rectal suction and full-thickness biopsy AchE Resection and pull through No
Rolle et al. [6] 6 Rectal suction and full-thickness biopsy AchE, NADPH, ICC, c-kit 5 Resection and pull through, 1 enterostomy No
Kobayashi et al. [2] 3 Full-thickness biopsy AchE Enterostomy All died
Kubota et al. [7] 6 Full-thickness biopsy S-100 Enterostomy All died
Meier-Ruge et al. [3] 7 Full-thickness biopsy AchE, LDH, SDH Resection and pull through No
Schaerli et al. [8] 7 Full-thickness biopsy AchE, LDH, SDH Resection and pull through No
Ure et al. [9] 9 Rectal suction biopsy AchE, LDH, SDH 7 resection and pull through, 2 sphincter myotomy 1 died
Yamataka et al. [10] 1 Full-thickness biopsy AchE, ICC, c-kit Enterostomy No
Munakata et al. [11] 12 Rectal suction and full-thickness biopsy AchE, silver impregnation 11 resection and pull through, 1 not reported No
Meier-Ruge [12] 18 Full-thickness biopsy AchE, LDH, SDH Not reported No
Munakata et al. [13] 6 Full-thickness biopsy AchE, silver impregnation Not reported No
  1. Adapted from Dingemann J, Puri P: Isolated hypoganglionosis: systematic review of a rare intestinal innervation defect. Pediatr Surg Int 26:1111-1115 [1] (Permission granted).
  2. AchE = acetylcholinesterase; ICC = interstitial cells of Cajal; LDH = lactate dehydrogenase; NADPH = nicotinamide adenine dinucleotide phosphate; SDH = succinate dehydrogenase.