Clinical Review & Education
JAMA Surgery Clinical Challenge
An Uncommon Surgical Disease Julien Jarry, MD; Thierry Peycru, MD; Manu Shekher, MD; Jean Luc Faucheron, MD, PhD
Figure 1. View of the mass exteriorized through the anus.
A woman in her 50s was hospitalized for a painful anal mass. She had a history of hyWHAT IS THE DIAGNOSIS?
pertension and depression and was receiving omeprazole and paroxetine. The mass had appeared 2 days before presentation and was associated with vomiting. Addition-
A. Hedrocele
ally, the patient had not passed stools or had intestinal gas for 2 days. On physical exB. Rectal prolapse
amination, the mass was exteriorized through the anal Quiz at jamasurgery.com
canal. It was covered by hypoxemic rectal mucosa, but
C. Anal neoplasm
no digestive lumen could be identified inside the mass D. External hemorrhoids
(Figure 1). Furthermore, the patient had a distended abdomen, absent bowel sounds, and no tenderness to palpation. No abdominal scar was visible, and no groin hernia was palpable. Her blood pressure was 160/80 mm Hg and her temperature was 37.8°C. Results of complete blood cell count, coagulation tests, and basic chemistry panel were all within normal limits. An abdominal radiograph showed several air-fluid levels without pneumoperitoneum, confirming an intestinal occlusion.
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JAMA Surgery April 2014 Volume 149, Number 4
Copyright 2014 American Medical Association. All rights reserved.
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395
Clinical Review & Education JAMA Surgery Clinical Challenge
Diagnosis A. Hedrocele
Discussion With the patient under brief general anesthesia, the mass was reintroduced into the anus and, to prevent recurrence, an anal probe was placed in the rectum (Figure 2). After intestinal transit resumed, the patient underwent static pelvic floor magnetic resonance imaging, which confirmed the diagnosis of hedrocele (Figure 3). In view of these findings, the diagnosis of hedrocele complicated by an intestinal incarceration was made. The patient underwent an operation 6 days later. A laparoscopic rectopexy was performed, and the patient was discharged home on the fifth postoperative day. At 2-years’ follow-up, the patient is well, without recurrence of the hedrocele. Hedrocele, derived from the Greek terms hedros, meaning anus, and kele, meaning hernia, literally means anal hernia. It represents an extremely rare form of hernia—only 2 case reports1,2 have been published in the medical literature. Like enterocele, hedrocele is a variant of the rare posterior perineal hernia, resulting from a defect in the rectovaginal septum.3 The herniation resulting from this defect can protrudeanteriorlyorposteriorly.Inwomen,theherniastypicallyprotrude in the posterior wall of the vagina and are termed enteroceles; in men they protrude posteriorly in the anterior wall of the rectum and are called hedroceles. This difference is due to the resistance of the prostate gland in men, which tends to favor posterior protrusion. However, hedrocele can occur in women, as illustrated in our case. It can be associated with enterocele or other perineal hernias. From a clinical point of view, hedrocele can cause different but nonspecific symptoms, such as dyschezia, constipation, and tenesmus. The natural history of hedrocele is to progress and protrude through the rectum and then through the anus. The main complication is intestinal incarceration and strangulation in the hernia. The differential diagnosis of hedrocele includes rectal polyps, rectal tumors, rectal prolapse, and hemorrhoids. Differentradiologicexaminationscanhelptodiagnosehedrocele,such as defecography associated with bowel opacification. However, pelvicfloormagneticresonanceimagingiscurrentlythecriterionstandard to visualize hedrocele.4 The recommended treatment of hedrocele is rectopexy, which ideally can be performed under laparoscopy.5
U
P
V R
Figure 3. Pelvic magnetic resonance image showing the hedrocele (H) containing intestinal loops protruding into the rectum (R). B indicates bladder; P, pubis; U, uterus; and V, vagina.
Accepted for Publication: September 24, 2012.
Author Affiliations: Department of Digestive Surgery, Military Hospital Desgenettes, Lyon, France (Jarry); Department of Digestive Surgery, Military Hospital Robert Picqué, Bordeaux, France (Peycru); Emergency Medicine, St Vincent's Medical Center, Bridgeport, Connecticut (Shekher); Department of Colorectal Surgery, University Hospital, Grenoble, France (Faucheron).
Published Online: February 26, 2014. doi:10.1001/jamasurg.2013.808.
Section Editor: Carl E. Bredenberg, MD.
B H
ARTICLE INFORMATION
Corresponding Author: Julien Jarry, MD, Department of Digestive Surgery, Military Hospital Desgenettes, 69275 Lyon, France ([email protected]).
396
Figure 2. View of the rectal probe after the mass was reintroduced into the anus.
3. Stamatiou D, Skandalakis JE, Skandalakis LJ, Mirilas P. Perineal hernia. Am Surg. 2010;76(5):474-479.
REFERENCES
4. Macura KJ. Magnetic resonance imaging of pelvic floor defects in women. Top Magn Reson Imaging. 2006;17(6):417-426.
1. Maull KI, Fleishman HA. Hedrocele: report of a case and review of the literature. Dis Colon Rectum. 1978;21(2):107-109.
5. Salum MR, Prado-Kobata MH, Saad SS, Matos D. Primary perineal posterior hernia. Clinics (Sao Paulo). 2005;60(1):71-74.
Conflict of Interest Disclosures: None reported.
2. Mongardini M, Cola A, Iachetta RP, et al. Treatment of obstructive defecation syndrome related to hedrocele [in Italian]. G Chir. 2010;31(11-12):502-506.
JAMA Surgery April 2014 Volume 149, Number 4
Copyright 2014 American Medical Association. All rights reserved.
Downloaded From: http://archsurg.jamanetwork.com/ by a Nanyang Technological University User on 06/07/2015
jamasurgery.com
JAMA Surgery Clinical Challenge
An Uncommon Surgical Disease Julien Jarry, MD; Thierry Peycru, MD; Manu Shekher, MD; Jean Luc Faucheron, MD, PhD
Figure 1. View of the mass exteriorized through the anus.
A woman in her 50s was hospitalized for a painful anal mass. She had a history of hyWHAT IS THE DIAGNOSIS?
pertension and depression and was receiving omeprazole and paroxetine. The mass had appeared 2 days before presentation and was associated with vomiting. Addition-
A. Hedrocele
ally, the patient had not passed stools or had intestinal gas for 2 days. On physical exB. Rectal prolapse
amination, the mass was exteriorized through the anal Quiz at jamasurgery.com
canal. It was covered by hypoxemic rectal mucosa, but
C. Anal neoplasm
no digestive lumen could be identified inside the mass D. External hemorrhoids
(Figure 1). Furthermore, the patient had a distended abdomen, absent bowel sounds, and no tenderness to palpation. No abdominal scar was visible, and no groin hernia was palpable. Her blood pressure was 160/80 mm Hg and her temperature was 37.8°C. Results of complete blood cell count, coagulation tests, and basic chemistry panel were all within normal limits. An abdominal radiograph showed several air-fluid levels without pneumoperitoneum, confirming an intestinal occlusion.
jamasurgery.com
JAMA Surgery April 2014 Volume 149, Number 4
Copyright 2014 American Medical Association. All rights reserved.
Downloaded From: http://archsurg.jamanetwork.com/ by a Nanyang Technological University User on 06/07/2015
395
Clinical Review & Education JAMA Surgery Clinical Challenge
Diagnosis A. Hedrocele
Discussion With the patient under brief general anesthesia, the mass was reintroduced into the anus and, to prevent recurrence, an anal probe was placed in the rectum (Figure 2). After intestinal transit resumed, the patient underwent static pelvic floor magnetic resonance imaging, which confirmed the diagnosis of hedrocele (Figure 3). In view of these findings, the diagnosis of hedrocele complicated by an intestinal incarceration was made. The patient underwent an operation 6 days later. A laparoscopic rectopexy was performed, and the patient was discharged home on the fifth postoperative day. At 2-years’ follow-up, the patient is well, without recurrence of the hedrocele. Hedrocele, derived from the Greek terms hedros, meaning anus, and kele, meaning hernia, literally means anal hernia. It represents an extremely rare form of hernia—only 2 case reports1,2 have been published in the medical literature. Like enterocele, hedrocele is a variant of the rare posterior perineal hernia, resulting from a defect in the rectovaginal septum.3 The herniation resulting from this defect can protrudeanteriorlyorposteriorly.Inwomen,theherniastypicallyprotrude in the posterior wall of the vagina and are termed enteroceles; in men they protrude posteriorly in the anterior wall of the rectum and are called hedroceles. This difference is due to the resistance of the prostate gland in men, which tends to favor posterior protrusion. However, hedrocele can occur in women, as illustrated in our case. It can be associated with enterocele or other perineal hernias. From a clinical point of view, hedrocele can cause different but nonspecific symptoms, such as dyschezia, constipation, and tenesmus. The natural history of hedrocele is to progress and protrude through the rectum and then through the anus. The main complication is intestinal incarceration and strangulation in the hernia. The differential diagnosis of hedrocele includes rectal polyps, rectal tumors, rectal prolapse, and hemorrhoids. Differentradiologicexaminationscanhelptodiagnosehedrocele,such as defecography associated with bowel opacification. However, pelvicfloormagneticresonanceimagingiscurrentlythecriterionstandard to visualize hedrocele.4 The recommended treatment of hedrocele is rectopexy, which ideally can be performed under laparoscopy.5
U
P
V R
Figure 3. Pelvic magnetic resonance image showing the hedrocele (H) containing intestinal loops protruding into the rectum (R). B indicates bladder; P, pubis; U, uterus; and V, vagina.
Accepted for Publication: September 24, 2012.
Author Affiliations: Department of Digestive Surgery, Military Hospital Desgenettes, Lyon, France (Jarry); Department of Digestive Surgery, Military Hospital Robert Picqué, Bordeaux, France (Peycru); Emergency Medicine, St Vincent's Medical Center, Bridgeport, Connecticut (Shekher); Department of Colorectal Surgery, University Hospital, Grenoble, France (Faucheron).
Published Online: February 26, 2014. doi:10.1001/jamasurg.2013.808.
Section Editor: Carl E. Bredenberg, MD.
B H
ARTICLE INFORMATION
Corresponding Author: Julien Jarry, MD, Department of Digestive Surgery, Military Hospital Desgenettes, 69275 Lyon, France ([email protected]).
396
Figure 2. View of the rectal probe after the mass was reintroduced into the anus.
3. Stamatiou D, Skandalakis JE, Skandalakis LJ, Mirilas P. Perineal hernia. Am Surg. 2010;76(5):474-479.
REFERENCES
4. Macura KJ. Magnetic resonance imaging of pelvic floor defects in women. Top Magn Reson Imaging. 2006;17(6):417-426.
1. Maull KI, Fleishman HA. Hedrocele: report of a case and review of the literature. Dis Colon Rectum. 1978;21(2):107-109.
5. Salum MR, Prado-Kobata MH, Saad SS, Matos D. Primary perineal posterior hernia. Clinics (Sao Paulo). 2005;60(1):71-74.
Conflict of Interest Disclosures: None reported.
2. Mongardini M, Cola A, Iachetta RP, et al. Treatment of obstructive defecation syndrome related to hedrocele [in Italian]. G Chir. 2010;31(11-12):502-506.
JAMA Surgery April 2014 Volume 149, Number 4
Copyright 2014 American Medical Association. All rights reserved.
Downloaded From: http://archsurg.jamanetwork.com/ by a Nanyang Technological University User on 06/07/2015
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