A 12-year-old Girl presented to paediatric ED at night with acute onset of abdominal pain and persistent vomiting for the past eight hours. There was no blood, mucous or bile stain in her vomit. She was unable to tolerate anything orally and had persistent abdominal pain. She was triaged as category 3, and was reviewed by the paediatric team initially, for vomiting and abdominal pain in the paediatric ED. 

She had her all vaccinations and had an allergy to amoxicillin, with a history of rashes in the past. She was in main stream school, with normal developmental history but there were some concerns of Asperger’s syndrome, for which she was awaiting an assessment. She was vitally stable on presentation with a heart rate of 105, BP: 114/77, respiratory rate: 22 and she was afebrile.

On examination two hours after presentation, her condition had worsened; she was weak, lethargic and her appearance was pale with a capillary refill time of two to three seconds, and dry mucous membrane. She had tachycardia with normal BP. She had shallow breathing with marked abdominal distention generalised tenderness and positive bowel sounds. Other systemic examination was unremarkable. The child was also noted to have a high pain threshold on examination. Clinically she was noted to have acute abdomen pain, likely due to a large bowel obstruction or volvulus.

Investigations

This patient was started on intravenous fluids bolus and given triple antibiotics after obtaining baseline bloods including FBC, U&E, LFT, amylase, CRP and VBG and blood culture. She was kept NPO and the surgical team was contacted for urgent review. Her VBG had pH: 7.35, pCo2: 4.9, pO2: 9.8, glucose: 10.9, lactate: 2.1, WBC: 3.7, neuro: 2.59, lymph: 1.05, HB: 13.6, CRP: 5, normal electrolytes and liver functions. 

Abdominal x-ray obtained after two hours of presentation showed dilated large bowel loops at uneven areas, which suggested acute intestinal obstruction or volvulus. The patient was then reviewed by the surgical team. A CT abdomen was preformed, which showed marked distension and filling of the stomach with heterogeneous material, suggestive of gastrointestinal (GI) bezoars. There was evidence of acute hollow organ perforation with large amount of free fluid and free air in the abdominal cavity. She was transferred urgently to tertiary care for open laparotomy.

Open laparotomy was performed on the same day with almost 1kg (sugar bag size) of hairs found in her stomach. Resection anastomosis repair was carried out for the perforation, the patient needed inotropes in ICU on day two of surgery and was moved to a ward on day five post surgery. She spent three weeks in hospital and was later discharged home.

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Retrospectively her mother said that she noticed broken hairs on her child’s head for past three to four years, but never had a suspicion of trichotillomania or trichophagia, nor did she think that she would end up with such a surgical emergency.

Discussion

Trichotillomania is a chronic, mental health disease of impulse control, characterised by repetitive, compulsive  and self-induced hair pulling. It can occur at any age but is observed more often in adolescents, with a strong predominance in females.¹

Diagnosis of trichotillomania may be difficult, and its effective treatment is challenging. Trichotillomania is often connected with trichophagia, which may lead to formation of trichobezoars and cause a direct danger to a person’s health and even life, due to the risk of intestinal obstruction and the need for surgical intervention.

The term ‘trichotillomania’ was first used by Hallopeau in 1889. It originates from the Greek words ‘thrix’ (hair), ‘tillein’ (to pull) and ‘mania’ (madness).²

A bezoar is a tightly packed collection of partially digested or undigested material, mainly hairs, that most commonly occurs in the stomach. Different types of bezoars include: phytobezoar (vegetable origin), trichobezoar (composed mainly of hair), lactobezoar (concentrated milk formula), pharmacobezoar (mixed medicine bezoar), and food-bolus bezoars.³ Trichobezoars (concretions of hair) are unusual and are usually found in young females with a psychiatric condition, who often deny eating their own hair.

Gastric bezoars can occur in all age groups and often occur in patients with behaviour disorders, abnormal gastric emptying or altered gastrointestinal anatomy. Although the prevalence of bezoars in humans is low, an absence of treatment has been associated with mortality rates as high as 30%, primarily because of gastrointestinal bleeding, destruction or perforation.⁴ In some cases, however, a trichobezoar extends through the pylorus into jejunum, ileum or even colon. This condition, called Rapunzel syndrome, was first described by Vaughan et al in 1968.⁵

Bezoars may present with abdominal pain, nausea/vomiting, early satiety, weight loss, intestinal obstruction and ulceration leading to bleeding and/or perforation. In this case almost all of the acute symptoms were present on arrival to ED, except the chronic hair pulling or hair eating history.

Conclusion

Although rare, trichobezoars should not be forgotten as a differential diagnosis in young females presenting with acute abdominal pain and vomiting. The earlier the diagnosis, the quicker the resolution and avoidance of complications. In addition, recurrences are known. A psychiatric evaluation, counselling and treatment are helpful in preventing these recurrences.

References

1. Cisoń H, Kuś A, Popowicz E, Szyca M, Reich A. Trichotillomania and trichophagia: modern diagnostic and therapeutic methods. Dermatol Ther  2018 Sep; 8(3):389-98. doi: 10.1007/s13555-018-0256-z
2. Parakh P, Srivastava M. The many faces of trichotillomania. Int J Trichology 2010 Jan-Jun; 2(1):50-2. doi: 10.4103/0974-7753.66916
3. Iwamuro M, Okada H, Matsueda K et al. Review of the diagnosis and management of gastrointestinal bezoars. World J Gastrointest Endosc 2015 Apr 16; 7(4):336-45. doi: 10.4253/wjge.v7.i4.336
4. Eng K, Kay M. Gastrointestinal bezoars: history and current treatment paradigms. Gastroenterol Hepatol 2012 Nov; 8(11):776-8. PMC3966178/
5. Lopes LR, Oliveira PS, Pracucho EM et al. The Rapunzel syndrome: an unusual trichobezoar presentation. Case Rep Med 2010; 2010:841028. doi:10.1155/2010/841028