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      A Boy Who Knows No Pain: Anaesthetic Management of Congenital Insensitivity to Pain With Anhidrosis

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      Publication date (Electronic, pub):
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      Journal: Cureus
      Publisher: Cureus
      Keywords: cipa, analgesia, anaesthetic complications, perioperative management, autonomic dysregulation

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          Abstract

          Congenital insensitivity to pain with anhidrosis (CIPA) is a rare disorder with an absence of pain perception, anhidrosis, heat intolerance, and varying degrees of mental retardation. Though cases of CIPA have innate analgesia, they have been known to have tactile hyperesthesia, thus making anesthesia necessary in case of any surgery. Perioperative complications due to abnormal autonomic functions like bradycardia, hypotension, and hyperthermia are major challenges in the anesthetic management of these cases. Here, we report a case on the anesthetic management of CIPA.

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          Update Review and Clinical Presentation in Congenital Insensitivity to Pain and Anhidrosis

          L. M. Pérez-López, M. Cabrera-González, D. Gutiérrez-de la Iglesia … (2015)
          Introduction. Congenital insensitivity to pain and anhidrosis (CIPA) or hereditary sensory and autonomic neuropathy type IV is an extremely rare syndrome. Three clinical findings define the syndrome: insensitivity to pain, impossibility to sweat, and mental retardation. This pathology is caused by a genetic mutation in the NTRK1 gene, which encodes a tyrosine receptor (TrkA) for nerve growth factor (NGF). Methods. The consultation of a child female in our center with CIPA and a tibia fracture in pseudoarthrosis encouraged us to carefully review literature and examine the therapeutic possibilities. A thorough review of literature published in Pubmed was done about CIPA and other connected medical issues mentioned in the paper. Conclusions. The therapeutic approach of CIPA remains unclear. The preventive approach remains the only possible treatment of CIPA. We propose two new important concepts in the therapeutic approach for these patients: (1) early surgical treatment for long bone fractures to prevent pseudoarthrosis and to allow early weight bearing, decreasing the risk of further osteopenia, and (2) bisphosphonates to avoid the progression of osteopenia and to reduce the number of consecutive fractures.
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            Congenital Insensitivity to Pain and Anhydrosis (CIPA) Syndrome; A Report of 4 Cases

            Khadije Daneshjou, Hanieh Jafarieh, Seyed-Reza Raaeskarami (2012)
            Background Background: Congenital insensitivity to pain with anhidrosis (CIPA) is characterized by recurrent episodes of infections and unexplained fever, anhidrosis (inability to sweat), and absence of reaction to noxious stimuli, self-mutilating behavior, mental retardation and damages to oral structures. Case Presentation In this article, we have demonstrated the signs and symptoms of 4 children that refer to the pediatrics department of the Imam Khomeini hospital and assay about their complications with this disease. They mostly presented by recurrent osteomyelitis in their feet that severely controlled by antibiotic therapy and even surgery. They had no pain sensation in spite of deep sore and infection. Conclusion This syndrome can be diagnosed by clinical and paraclinical tests together but it would be better to confirm by genetic test. The diagnosis of this syndrome helps us to try for the better quality of life for the patients and avoid unnecessary amputations.
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              Anesthetic Management of Patients with Congenital Insensitivity to Pain with Anhidrosis: A Retrospective Analysis of 358 Procedures Performed Under General Anesthesia.

              Alexander Zlotnik, Dmitry Natanel, Ruslan Kutz … (2015)
              Congenital insensitivity to pain with anhidrosis (CIPA) is a rare autosomal recessive disorder characterized by recurrent episodic fevers, anhidrosis, absent reaction to noxious stimuli, self-mutilating behavior, and mental retardation. The anesthetic management of patients with CIPA is challenging. Autonomic nervous system abnormalities are common, and patients are at increased risk for perioperative complications.
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                Author and article information

                Journal
                Journal ID (nlm-ta): Cureus
                Journal ID (iso-abbrev): Cureus
                Journal ID (issn): 2168-8184
                Title: Cureus
                Publisher: Cureus (Palo Alto (CA) )
                ISSN (Electronic): 2168-8184
                Publication date (Electronic, pub): 28 October 2022
                Publication date (Electronic, collection): October 2022
                Volume: 14
                Issue: 10
                Electronic Location Identifier: e30790
                Affiliations
                [1 ] Anesthesia and Critical Care, Vardhman Mahavir Medical College and Safdarjung Hospital, New Delhi, IND
                Author notes
                Article
                DOI: 10.7759/cureus.30790
                PMC ID: 9701320
                PubMed ID: 36447712
                SO-VID: 3930ca38-0d46-4dc9-8c78-a8d89e0d45cd
                Copyright © Copyright © 2022, Paul et al.
                License:

                This is an open access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited.

                History
                Date accepted : 28 October 2022
                Categories
                Subject: Anesthesiology
                Subject: Pain Management
                Subject: Orthopedics

                Keywords: cipa,analgesia,anaesthetic complications,perioperative management,autonomic dysregulation
                Data availability:
                Keywords: cipa, analgesia, anaesthetic complications, perioperative management, autonomic dysregulation

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