Pneumatosis intestinalis in a patient with trichobezoar – rare association
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1) A 17-year-old girl presented with chronic epigastric pain and was found to have a large trichobezoar in her stomach. 2) During surgery to remove the bezoar, the doctors also found extensive pneumatosis intestinalis (intramural gas within the bowel walls) in her small intestine. 3) This case presents a rare association between trichobezoar and pneumatosis intestinalis that has not been previously reported. As the pneumatosis was not causing symptoms, no treatment was needed for it.
![Creative Commons 4.0
Pneumatosis intestinalis in a patient with trichobezoar – Rare
association
Varuna B. Pathirana a,*
, Rakitha V. Paranamanna a
, Gayan R. M. Rathnayaka a
, Mahanama. Gunasekara a
a Department of General Surgery, National Hospital, Colombo, Sri Lanka.
* Corresponding author: Varuna Bhagya Pathirana
Mailing address: Department of General Surgery, Ward 32, Na-
tional Hospital, Colombo, Sri Lanka.
E-mail: varunapath@gmail.com
Received: 29 December 2020 / Accepted: 28 January 2021
INTRODUCTION
Trichobezoar is a mass of ingested hair within the gas-
trointestinal tract [1]
. This rare cause was found to be
the reason for chronic epigastric pain of a young girl
in this case report without apparent psychiatric ill-
ness. She was incidentally found to have pneumatosis
intestinalis (PI) which may be or may not be related to
trichobezoar [2]
.
CASE REPORT
A 17-year-old girl presented with epigastric pain for
one-year duration associated with nausea, postprandi-
al abdominal fullness, early satiety, and episodic vom-
iting occurring 3-4 times a month. Her bowel habits
were normal and there was no per rectal bleeding or
melena. She was admitted to the hospital due to wors-
ening of symptoms over two weeks with more frequent
vomiting. She denies trichotillomania and there was no
Case Report
history of psychiatric illness or behavioral abnormality
and her school performances were average.
She was not pale, anicteric, BMI was 17.18 kg/m2
and
no abnormality was noted in her hair. Abdominal ex-
amination revealed a firm lump involving epigastric
and right hypochondria extending 6 cm from the costal
margin. The rest of the abdominal examination was
unremarkable. Her hemoglobin was 12.7 g/dl, serum
albumin was 4.0 g/dl and the rest of the laboratory in-
vestigations, including bilirubin levels, serum amylase,
and serum electrolytes were within the normal range.
Gastroduodenoscopy revealed large trichobezoar
where the scope was not negotiable beyond the body
of the stomach (Figure 1). CECT showed grossly dis-
tended stomach with non-enhancing intraluminal mass
extending up to the first part of duodenum suggestive
of a bezoar (Figure 2) and intramural gas in the small
and large intestine suggestive of pneumatosis.
She underwent laparotomy and the bezoar was re-
trieved via gastrotomy (Figure 3). A gastric wall thick-
ening was observed with a thickness of 5-6 mm. Distal
jejunum and entire ileum showed gaseous outpouchings
of variable size ranging from few millimeters to 1 cm in-
dicating extensive intestinal Pneumatosis (Figure 4) and
was left unattended since the patient was asymptom-
atic. She made the uneventful postoperative recovery.
Abstract
This case report describes a young girl who presented with chronic epigastric pain and abdominal mass
without noticeable psychiatric illness or trichotillomania and subsequently trichobezoar was found to be the
reason for her symptoms. She underwent laparotomy to retrieve the bezoar. During laparotomy extensive
pneumatosis of the small bowel was noted where this association was not previously reported in the literature.
Since pneumatosis was not symptomatic no bowel resection was carried out. She made an uneventful recovery.
This illustrates that trichobezoar is an important cause to consider in young females with chronic abdominal
pain even in the absence of clear evidence for trichotillomania. Rarely, this can be associated with intestinal
pneumatosis. Intestinal pneumatosis does not warrant treatment unless it causes symptoms.
Keywords: Trichobezoar; epigastric pain; pneumatosis intestinalis; trichotillomania; pneumoperitoneum
V. B. Pathirana et al 18
Clin Surg Res Commun 2020; 5(1): 18-21
DOI: 10.31491/CSRC.2021.03.069](https://image.slidesharecdn.com/pneumatosisintestinalisinapatientwithtrichobezoarrareassociation-210819062034/85/Pneumatosis-intestinalis-in-a-patient-with-trichobezoar-rare-association-1-320.jpg)
![She was referred to a psychiatrist on discharge from the
hospital and no active treatment was recommended.
DISCUSSION
Bezoars are masses formed from ingested foreign ma-
terial and identified in less than 5% of all upper gas-
trointestinal endoscopy [1]
. They are most commonly
found in the stomach. Trichobezoar is the rarest of
them accounting for 6% of all bezoars and invariably
related to trichotillomania and trichophagia [1]
which
can be a part of a psychiatric illness. Even though the
presence of trichobezoar is a piece of clear evidence
for trichophagia, some patients may deny these habits
similar to this case.
Bezoars can be asymptomatic particularly when they
are small and symptomatic patients have epigastric
pain, nausea, and vomiting, early satiety which is non-
specific and can be overlooked as young patients may
not undergo routine endoscopy leading to delay of
diagnosis. Because around 90% of these patients are
young females [1,4]
, this case illustrates the importance
of considering trichobezoar as a cause for unexplained
abdominal pain in a young female even with the ab-
sence of clear psychiatric illness or evidence for tricho-
tillomania.
Trichobezoar needs early intervention since it is
known for many complications such as gastric erosion
and bleeding, perforation, and gastric outlet obstruc-
tion. The extension of trichobezoar into the small bow-
el which is known as Rapunzel Syndrome can lead to
small bowel obstruction, pancreatitis, and cholangitis
[1,3,5]
. Open surgery is considered the best method since
it is technically easy and has a high success rate of
complete removal of the bezoar with low complication
rates [1,3,5]
. Laparoscopic and endoscopic interventions
are shown to be less effective [1,3,5]
. Most patients will
only need gastrotomy and/or enterotomy for retrieval
of bezoar, but complicated cases may require subtotal
gastrectomy or intestinal resection [1]
. Following the
successful removal of trichobezoar patients will need
surveillance endoscopy for few years to confirm the
patient has come out of trichotillomania [3]
.
Pneumatosis intestinalis (PI) is a rare condition that
has a wide range of clinical presentations from benign
asymptomatic condition to a life-threatening surgical
emergency due to adhesion obstruction or spontane-
ous bowel perforation [2,6]
. Morphologically PI is seen
in “bubbles like” type (cystoides) which was noted in
this patient or a continuous band-like type [2]
. It may be
idiopathic (primary), occasionally identified in asymp-
tomatic patients, or secondary to various gastrointes-
tinal or pulmonary diseases [2,7]
. Inflammatory bowel
disease, chronic bowel infections and infestations,
bowel obstruction and pyloric obstruction, diverticu-
litis, bowel ischemia, toxic megacolon are some of the
common gastrointestinal conditions related to PI [7,8]
.
Some of the non-gastrointestinal conditions related
to PI are pulmonary diseases like asthma, and emphy-
sema, collagen vascular diseases, immunosuppression,
organ transplantation ect [7,8]
. Direct association with
trichobezoar and PI is not documented in the litera-
ture. Chronic inflammation of the bowel wall second-
ary to hair, partial pyloric, and small bowel obstruction
may have contributed to pneumatosis in this patient
since these causes are known to associate with PI [7]
.
Most of the time PI will present with symptoms related
V. B. Pathirana et al 19
Figure 1. Trichobezoar.
Figure 2. Heterogeneous mass within the stomach.
ANT PUBLISHING CORPORATION
Published online: 29 March 2021](https://image.slidesharecdn.com/pneumatosisintestinalisinapatientwithtrichobezoarrareassociation-210819062034/85/Pneumatosis-intestinalis-in-a-patient-with-trichobezoar-rare-association-2-320.jpg)
![ration or obstruction. Patients with clinical and labo-
ratory evidence of abdominal sepsis elevated serum
amylase, and presence of portal venous gas need ur-
gent surgery where asymptomatic cases are managed
non operatively [2,6,8]
. Pneumoperitoneum alone should
not be considered as an indication for surgery since it
was noted in 50% of patients with PI [6]
. In this patient
resection of the involved segment was not necessary
as it is uncomplicated and neither feasible considering
the extensive involvement of the small bowel.
CONCLUSION
Trichobezoar can be a cause for chronic unexplained
abdominal pain in young females, where it may be not
suspected in the absence of trichotillomania and hair
loss. Trichobezoar and intestinal pneumatosis are rare
clinical entities where this association was not docu-
mented previously. Pneumatosis intestinalis needs in-
dividualized treatment based on clinical presentation.
DECLARATIONS
Authors’ contributions
All authors made substantial contributions to merit
inclusion as co-authors. All authors approved the final
manuscript.
Conflict of interest
All authors declared that there are no conflicts of inter-
est.
Ethics approval
Not applicable.
Consent for publication
Patient consent was obtained for publication without
personal details.
REFERENCES
1. García-Ramírez, B. E., Nuño-Guzmán, C. M., Zaragoza-Car-
rillo, R. E., Salado-Rentería, H., Gómez-Abarca, A., & Co-
rona, J. L. (2018). Small-bowel obstruction secondary to
ileal trichobezoar in a patient with Rapunzel syndrome.
Case reports in gastroenterology, 12(3), 559-565.
2. Berritto, D., Crincoli, R., Iacobellis, F., Iasiello, F., Pizza, N.
L., Lassandro, F., ... & Grassi, R. (2014). Primary pneuma-
tosis intestinalis of small bowel: a case of a rare disease.
Case reports in surgery, 2014.
3. Wolski, M., Gawłowska-Sawosz, M., Gogolewski, M.,
Wolańczyk, T., Albrecht, P., & Kamiński, A. (2016). Tricho-
tillomania, trichophagia, trichobezoar-summary of three
cases. Endoscopic follow up scheme in trichotillomania.
Clin Surg Res Commun 2020; 5(1): 18-21
DOI: 10.31491/CSRC.2021.03.069
V. B. Pathirana et al 20
to its underlying disease. However, vomiting, abdomi-
nal distention, weight loss with the involvement of the
small bowel and diarrhea, hematochezia with the in-
volvement of the large bowel is reported as the symp-
toms related to PI [8]
.
A plain abdominal x-ray may reveal pneumatosis but
a contrast-enhanced CT scan of the abdomen is more
sensitive in detecting PI and particularly in acute pre-
sentation it can help in identifying Intraluminal gas
from intramural gas, the air within the biliary system,
ischemic bowel, pneumoperitoneum, ascites which will
help in the decision to ope rate [2,6,7]
.
Pneumatosis intestinalis is treated according to the
underlying cause and surgery is usually reserved for
patients presenting with acute abdomen due to perfo-
Figure 3. Retrieved trichobezoar.
Figure 4. Distal jejunum and Ileum showing Pneumatosis
intestinalis.](https://image.slidesharecdn.com/pneumatosisintestinalisinapatientwithtrichobezoarrareassociation-210819062034/85/Pneumatosis-intestinalis-in-a-patient-with-trichobezoar-rare-association-3-320.jpg)
Pneumatosis intestinalis in a patient with trichobezoar – rare association
- 1. Creative Commons 4.0 Pneumatosis intestinalis in a patient with trichobezoar – Rare association Varuna B. Pathirana a,* , Rakitha V. Paranamanna a , Gayan R. M. Rathnayaka a , Mahanama. Gunasekara a a Department of General Surgery, National Hospital, Colombo, Sri Lanka. * Corresponding author: Varuna Bhagya Pathirana Mailing address: Department of General Surgery, Ward 32, Na- tional Hospital, Colombo, Sri Lanka. E-mail: [email protected] Received: 29 December 2020 / Accepted: 28 January 2021 INTRODUCTION Trichobezoar is a mass of ingested hair within the gas- trointestinal tract [1] . This rare cause was found to be the reason for chronic epigastric pain of a young girl in this case report without apparent psychiatric ill- ness. She was incidentally found to have pneumatosis intestinalis (PI) which may be or may not be related to trichobezoar [2] . CASE REPORT A 17-year-old girl presented with epigastric pain for one-year duration associated with nausea, postprandi- al abdominal fullness, early satiety, and episodic vom- iting occurring 3-4 times a month. Her bowel habits were normal and there was no per rectal bleeding or melena. She was admitted to the hospital due to wors- ening of symptoms over two weeks with more frequent vomiting. She denies trichotillomania and there was no Case Report history of psychiatric illness or behavioral abnormality and her school performances were average. She was not pale, anicteric, BMI was 17.18 kg/m2 and no abnormality was noted in her hair. Abdominal ex- amination revealed a firm lump involving epigastric and right hypochondria extending 6 cm from the costal margin. The rest of the abdominal examination was unremarkable. Her hemoglobin was 12.7 g/dl, serum albumin was 4.0 g/dl and the rest of the laboratory in- vestigations, including bilirubin levels, serum amylase, and serum electrolytes were within the normal range. Gastroduodenoscopy revealed large trichobezoar where the scope was not negotiable beyond the body of the stomach (Figure 1). CECT showed grossly dis- tended stomach with non-enhancing intraluminal mass extending up to the first part of duodenum suggestive of a bezoar (Figure 2) and intramural gas in the small and large intestine suggestive of pneumatosis. She underwent laparotomy and the bezoar was re- trieved via gastrotomy (Figure 3). A gastric wall thick- ening was observed with a thickness of 5-6 mm. Distal jejunum and entire ileum showed gaseous outpouchings of variable size ranging from few millimeters to 1 cm in- dicating extensive intestinal Pneumatosis (Figure 4) and was left unattended since the patient was asymptom- atic. She made the uneventful postoperative recovery. Abstract This case report describes a young girl who presented with chronic epigastric pain and abdominal mass without noticeable psychiatric illness or trichotillomania and subsequently trichobezoar was found to be the reason for her symptoms. She underwent laparotomy to retrieve the bezoar. During laparotomy extensive pneumatosis of the small bowel was noted where this association was not previously reported in the literature. Since pneumatosis was not symptomatic no bowel resection was carried out. She made an uneventful recovery. This illustrates that trichobezoar is an important cause to consider in young females with chronic abdominal pain even in the absence of clear evidence for trichotillomania. Rarely, this can be associated with intestinal pneumatosis. Intestinal pneumatosis does not warrant treatment unless it causes symptoms. Keywords: Trichobezoar; epigastric pain; pneumatosis intestinalis; trichotillomania; pneumoperitoneum V. B. Pathirana et al 18 Clin Surg Res Commun 2020; 5(1): 18-21 DOI: 10.31491/CSRC.2021.03.069
- 2. She was referred to a psychiatrist on discharge from the hospital and no active treatment was recommended. DISCUSSION Bezoars are masses formed from ingested foreign ma- terial and identified in less than 5% of all upper gas- trointestinal endoscopy [1] . They are most commonly found in the stomach. Trichobezoar is the rarest of them accounting for 6% of all bezoars and invariably related to trichotillomania and trichophagia [1] which can be a part of a psychiatric illness. Even though the presence of trichobezoar is a piece of clear evidence for trichophagia, some patients may deny these habits similar to this case. Bezoars can be asymptomatic particularly when they are small and symptomatic patients have epigastric pain, nausea, and vomiting, early satiety which is non- specific and can be overlooked as young patients may not undergo routine endoscopy leading to delay of diagnosis. Because around 90% of these patients are young females [1,4] , this case illustrates the importance of considering trichobezoar as a cause for unexplained abdominal pain in a young female even with the ab- sence of clear psychiatric illness or evidence for tricho- tillomania. Trichobezoar needs early intervention since it is known for many complications such as gastric erosion and bleeding, perforation, and gastric outlet obstruc- tion. The extension of trichobezoar into the small bow- el which is known as Rapunzel Syndrome can lead to small bowel obstruction, pancreatitis, and cholangitis [1,3,5] . Open surgery is considered the best method since it is technically easy and has a high success rate of complete removal of the bezoar with low complication rates [1,3,5] . Laparoscopic and endoscopic interventions are shown to be less effective [1,3,5] . Most patients will only need gastrotomy and/or enterotomy for retrieval of bezoar, but complicated cases may require subtotal gastrectomy or intestinal resection [1] . Following the successful removal of trichobezoar patients will need surveillance endoscopy for few years to confirm the patient has come out of trichotillomania [3] . Pneumatosis intestinalis (PI) is a rare condition that has a wide range of clinical presentations from benign asymptomatic condition to a life-threatening surgical emergency due to adhesion obstruction or spontane- ous bowel perforation [2,6] . Morphologically PI is seen in “bubbles like” type (cystoides) which was noted in this patient or a continuous band-like type [2] . It may be idiopathic (primary), occasionally identified in asymp- tomatic patients, or secondary to various gastrointes- tinal or pulmonary diseases [2,7] . Inflammatory bowel disease, chronic bowel infections and infestations, bowel obstruction and pyloric obstruction, diverticu- litis, bowel ischemia, toxic megacolon are some of the common gastrointestinal conditions related to PI [7,8] . Some of the non-gastrointestinal conditions related to PI are pulmonary diseases like asthma, and emphy- sema, collagen vascular diseases, immunosuppression, organ transplantation ect [7,8] . Direct association with trichobezoar and PI is not documented in the litera- ture. Chronic inflammation of the bowel wall second- ary to hair, partial pyloric, and small bowel obstruction may have contributed to pneumatosis in this patient since these causes are known to associate with PI [7] . Most of the time PI will present with symptoms related V. B. Pathirana et al 19 Figure 1. Trichobezoar. Figure 2. Heterogeneous mass within the stomach. ANT PUBLISHING CORPORATION Published online: 29 March 2021
- 3. ration or obstruction. Patients with clinical and labo- ratory evidence of abdominal sepsis elevated serum amylase, and presence of portal venous gas need ur- gent surgery where asymptomatic cases are managed non operatively [2,6,8] . Pneumoperitoneum alone should not be considered as an indication for surgery since it was noted in 50% of patients with PI [6] . In this patient resection of the involved segment was not necessary as it is uncomplicated and neither feasible considering the extensive involvement of the small bowel. CONCLUSION Trichobezoar can be a cause for chronic unexplained abdominal pain in young females, where it may be not suspected in the absence of trichotillomania and hair loss. Trichobezoar and intestinal pneumatosis are rare clinical entities where this association was not docu- mented previously. Pneumatosis intestinalis needs in- dividualized treatment based on clinical presentation. DECLARATIONS Authors’ contributions All authors made substantial contributions to merit inclusion as co-authors. All authors approved the final manuscript. Conflict of interest All authors declared that there are no conflicts of inter- est. Ethics approval Not applicable. Consent for publication Patient consent was obtained for publication without personal details. REFERENCES 1. García-Ramírez, B. E., Nuño-Guzmán, C. M., Zaragoza-Car- rillo, R. E., Salado-Rentería, H., Gómez-Abarca, A., & Co- rona, J. L. (2018). Small-bowel obstruction secondary to ileal trichobezoar in a patient with Rapunzel syndrome. Case reports in gastroenterology, 12(3), 559-565. 2. Berritto, D., Crincoli, R., Iacobellis, F., Iasiello, F., Pizza, N. L., Lassandro, F., ... & Grassi, R. (2014). Primary pneuma- tosis intestinalis of small bowel: a case of a rare disease. Case reports in surgery, 2014. 3. Wolski, M., Gawłowska-Sawosz, M., Gogolewski, M., Wolańczyk, T., Albrecht, P., & Kamiński, A. (2016). Tricho- tillomania, trichophagia, trichobezoar-summary of three cases. Endoscopic follow up scheme in trichotillomania. Clin Surg Res Commun 2020; 5(1): 18-21 DOI: 10.31491/CSRC.2021.03.069 V. B. Pathirana et al 20 to its underlying disease. However, vomiting, abdomi- nal distention, weight loss with the involvement of the small bowel and diarrhea, hematochezia with the in- volvement of the large bowel is reported as the symp- toms related to PI [8] . A plain abdominal x-ray may reveal pneumatosis but a contrast-enhanced CT scan of the abdomen is more sensitive in detecting PI and particularly in acute pre- sentation it can help in identifying Intraluminal gas from intramural gas, the air within the biliary system, ischemic bowel, pneumoperitoneum, ascites which will help in the decision to ope rate [2,6,7] . Pneumatosis intestinalis is treated according to the underlying cause and surgery is usually reserved for patients presenting with acute abdomen due to perfo- Figure 3. Retrieved trichobezoar. Figure 4. Distal jejunum and Ileum showing Pneumatosis intestinalis.
- 4. approach to pneumatosis intestinalis: Factors affecting your management. International journal of surgery case reports, 6, 133-137. 7. Ho, L. M., Paulson, E. K., & Thompson, W. M. (2007). Pneu- matosis intestinalis in the adult: benign to life-threaten- ing causes. American Journal of Roentgenology, 188(6), 1604-1613. 8. Zhang, H., Jun, S. L., & Brennan, T. V. (2012). Pneumatosis intestinalis: not always a surgical indication. Case reports in surgery, 2012. Psychiatr Pol, 50(1), 145-52. 4. Dindyal, S., Bhuva, N. J., Ramdass, M. J., & Narayansingh, V. (2008). Trichobezoar presenting with the’comma sign’in Rapunzel Syndrome: a case report and literature review. Cases journal, 1(1), 1-4. 5. Gorter, R. R., Kneepkens, C. M. F., Mattens, E. C. J. L., Aron- son, D. C., & Heij, H. A. (2010). Management of trichobe- zoar: case report and literature review. Pediatric surgery international, 26(5), 457-463. 6. Tahiri, M., Levy, J., Alzaid, S., & Anderson, D. (2015). An V. B. Pathirana et al 21 ANT PUBLISHING CORPORATION Published online: 29March 2021