Abstract

Citation: Bharti V, din Dar ND, Gania AM, din Bhat AM. Bleeding per rectum as a rare presentation of obsessive-compulsive disorder in a patient with Kallmann syndrome: A case report. J Clin Images Med Case Rep. 2024; 5(2): 2832.

Introduction

Kallmann syndrome is also called idiopathic hypogonadotropic hypogonadism with anosmia, a genetic disorder with rare occurrence in humans characterized by delay/absence of signs of puberty with an absent/impaired sense of smell. Hypothalamic dysfunction is suggested to be aetiologically involved [1]. Underdeveloped genitals and absent olfactory bulbs were reported by Spanish pathologist Juan in the mid-50s of the 19th century [2]. A combination of anosmia and hypothalamic dysfunction may be associated with moderate-to-severe intellectual disability, congenital ichthyosis, ocular defects, unilateral renal hypoplasia or agenesis and different movement disorders of limbs [3]. Insufficient secretion or action of gonadotropinreleasing hormone, low levels of testosterone, estradiol or inappropriately normal serum levels of gonadotropins have been found responsible [4]. In males, it exhibits a lack of virilization, and phenotypes such as low developed muscle, gynoid habitus, sparse type body hair, high-tone voice, and poorly developed genitalia. In females, lack of secondary sexual characteristics like breast development, pubic hair and amenorrhea. clinical presentation may be variable in non-reproductive phenotypes [5,6]. Obsessive Compulsive Disorder is a common chronic psychiatric disorder believed to have chronic waxing and waning course with a prevalence of 3% [1]. It is associated with decreased quality of life and functional impairment [7,8]. OCD can be treated successfully but patients respond to treatment at variable rates [9,10]. More physical symptoms are prevalent in the OCD population than in the general population as far as literature is concerned [11,12]. Neuropsychiatric manifestations such as negative symptoms like anhedonia, blunted affect, apathy, depression and sexual dysfunction were reported [13,14]. Kallman syndrome may be accompanied by psychotic phenomena within the schizophrenia spectrum [15].

Case presentation

18 years old unmarried male student known case of Kallman syndrome from rural strata of the population reffered from tertiary care for psychiatric consultation. Patient was apparently all right 2 years back when his mother approached the tertiary care hospital complaining that her son is eating inedible things Gypsum (calcium sulphate dihydrate), patient was evaluated there and different investigations were ordered, CBC revealed features of anaemia and patient was treated as a case of iron deficiency anaemia. The patient was put on the treatment for a few months, although the symptoms stopped for a few months and then again the patient started eating gypsum powder. In the meantime her mother weighing the gravity of the matter took him again to the treating hospital where the patient was evaluated CBC reported no deviations from the normal range this time. CEMRI BRAIN revealed, “ENHANCING LESION INVOLVING the HYPOTHALAMUS AND INFUNDIBULAR STALK” (possibilities of Langerhans cell histiocytosis and germinoma). The patient was operated on for the suprasellar mass. Resection was done and histopathological examination of the mass revealed features of pituitary adenoma. In view of the persistence of symptoms, and bleeding per rectum, the patient was asked to undergo an ultrasonography abdomen that revealed subacute intestinal obstruction, colonscopy turned out to be diagnostic for Chron`s disease. The patient was operated and a colostomy was done, after two weeks of discharge from the hospital, the patient every half an hour went to the toilet, mother of the patient got more suspicious about the child’s behaviour. Mother stalked patiently through the journey to the toilet and saw her child and mother noticed blood-tinged stools. The surgeon was again consulted for the said symptoms, the surgeon noticed some injury marks in the anal region likely self-induced. The surgeon referred the patient to psychiatry OPD. The patient was evaluated, a detailed assessment of the patient revealed that the patient has a compulsive behaviour of insertion of a finger in the rectum feeling a sense of fullness after defecation, in addition to repetitive hand washing and fear of contamination. The patient was assessed on YBOCS SCALE to determine the severity of the disease. YBOCS revealed a score of 37 (severe). The patient was diagnosed as a Diagnosis of OBSESSIVE COMPULSIVE DISORDER. Both the options of treatment pharmacotherapy and psychotherapy were discussed with the patient. The patient opted for pharmacotherapy, fluvoxamine 50 mg once a day for a week and then 100 mg in the morning and 50 in the evening was given over a period of 4 weeks. The patient showed improvement in the behaviour of inserting the finger in the rectum, eating gypsum (calcium sulphate dihydrate), repetitive hand washing and fear of contamination. The score on Y-BOCS was reduced to 11 after 6 weeks. To date, the patient is under constant psychiatric follow-up.

Discussion

This case report describes a patient that met the diagnostic criteria for OCD as per DSM-5 [16]. The patient had a history of PICA, and anaemia, which led him to seek consultation from a medicine specialist, though after some weeks patient’s anaemia improved. But subsequently patient suffered from constipation and tenesmus-like sensation, anal pain, bleeding per rectum, mucus discharge per rectum, irritation, and restlessness due to distension of the abdomen. Symptoms described were considered as the prodromal symptoms preceding the full-blown picture of subacute intestinal obstruction. Visualizing bloodtinged stool by mother compelled mother to visit a surgeon. The surgeon examined the patient and suspected self-induced anal tears and then the patient was referred to a psychiatrist. The patient was evaluated and a detailed examination was done. The patient revealed repetitive washing, and cleaning, a feeling of incomplete emptying, compulsive manual removal of stool from the rectum, and compulsive use of a finger for evacuation was reported by the patient. The patient was diagnosed as a case of obsessive-compulsive disorder. The severity of obsessions and compulsions was assessed by YBOCS-Scale. The patient was assessed on the Y-BOCS scale at 4 weeks and 8 weeks. A significant reduction was seen. Above case highlights the importance of early referral, early diagnosis and the pivotal role of psychopharmacological treatment in neuropsychiatric manifestations of hormonal dysfunction that has not responded to different surgical and medical methods of treatment that can prevent the medical complication. Such case reports have not been reported from the Indian subcontinent that have been published.

Conclusion

Patients diagnosed with different medical and surgical morbidities not responding to the conventional methods of treatment should be suspected for the possibility of psychiatric disorder and screened for the same.

Recommendations

• Obsessive-compulsive disorder should always be considered if a patient presents with Non-responsive repetitive medical/physical complaints.

• Patients with hormonal disorders should be assessed for any comorbid psychiatric disorders, early intervention in such cases is likely to improve the outcome of medical/surgical problems.

Consent: Written informed consent was taken from the patient and his guardian. Privacy and confidentiality was maintained.

References

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